Springer Science and Business Media LLCActa Medica Okayama2192-44491512026A case of tubulointerstitial nephritis with infiltration of neutrophils and interleukin-17-positive cells associated with Behçet’s disease35ENNaruhikoUchidaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKeikoTanakaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesNatsukiKubotaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesTakayukiKatsuyamaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKatsuyukiTanabeDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesHaruhito A.UchidaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesBehçet’s disease (BD) is a non-infectious inflammatory condition characterized by neutrophilic infiltration. In addition to primary symptoms, including oral and genital ulcers, ocular involvement, and skin lesions, BD can also affect various organs. However, renal involvement, particularly in tubulointerstitial nephritis, has rarely been described. Herein, a rare case of acute tubulointerstitial nephritis in a patient clinically diagnosed with BD is reported. The renal lesion presented with other symptoms of BD and fever, and was considered to be BD-related due to the presence of neutrophilic infiltration and its responsiveness to BD-directed therapy. Alterations in T-helper (Th) 1, Th2, and Th17 cytokine profiles are associated with BD activity. Interleukin (IL)-17 plays a central role in neutrophil activation, and recent studies have demonstrated a strong correlation between IL-17A levels and BD activity. In the present case, elevated serum IL-17A levels and infiltration of IL-17A-positive cells into the renal tissue reflected an active phase of BD and a BD-associated renal lesion.No potential conflict of interest relevant to this article was reported.岡山大学大学院ヘルスシステム統合科学研究科Acta Medica Okayama2436-322762026Shared Decision Making における患者参加の諸相と課題の考察1725ENMihoYOSHIDAGraduate School of Interdisciplinary Science and Engineering in Health Systems Okayama University10.18926/interdisciplinary/70327This paper traces the historical development of decision-making models in healthcare while exploring the meaning and practical significance of “patient participation” within the shared decision-making (SDM) framework. SDM is a recommended approach to clinical decision-making that emphasizes mutual information sharing and deliberation between physicians and patients. Traditional models often assume that patients can clearly articulate their values, preferences, and treatment goals. However, in actual clinical settings, particularly in cases of serious illness or life-threatening situations, patients frequently face emotional distress and psychological burdens, which can hinder their active participation in decision-making and the expression of their preferences. Based on SDM theory and practice reports, this study argues that SDM should not be viewed merely as a process that promotes patient choice. Even when patients choose not to actively participate and ultimately delegate decisions to healthcare providers or family members, such a choice can represent autonomous decision-making if it arises through meaningful communication and mutual understanding. This perspective calls for a more comprehensive and flexible interpretation of patient participation in SDM practice.No potential conflict of interest relevant to this article was reported.岡山大学理学部地球科学科Acta Medica Okayama1340-741432120262018年7月5日〜7日の西日本豪雨における広域降水特性3344ENKuranoshinKATOFaculty of Education, Okayama UniversityKengoMATSUMOTOOkayama Gakugeikan High SchoolKazuoOTANITV Setouchi Broadcasting Co., LTD.10.18926/ESR/70297 Large-scale rainfall characteristics at the heavy rainfall event around the western Japan for 5–7 July 2018 were analyzed with use of the 10-mimute precipitation data at the surface meteorological observation stations of the Japan Meteorological Agency, and so on. In this case, the area with 3 days total precipitation of near or more than 300 mm was distributed widely from northern Kyushu to Shiga and Fukui Prefectures. As in the many heavy rainfall events around Kyushu District in the mature stage of the Baiu season, contribution of the intense rainfall with more than 4 mm/10-minute (24 mm/h) attained about one third of the areal mean total precipitation. However, it is noted that the "not so intense rain" with less than 2 mm/10-minute (12 mm/h) also contributed to about one third of the huge total precipitation in the wide area. In short, this case could be characterized by the mixture of the western Japan type heavy rainfall event and the eastern Japan type one.No potential conflict of interest relevant to this article was reported.岡山大学理学部地球科学科Acta Medica Okayama1340-7414321202610分間降水量から大雨の特徴の多様性を捉える大学での授業の試み（防災気象リテラシー育成へ向けて）2131ENKuranoshinKATOFaculty of Education, Okayama University10.18926/ESR/70296 In the disaster prevention education on the heavy rainfall around Japan, it is also important to promote the meteorological literacy on the seasonal and regional differences of their rainfall characteristics such as the convective rain or stratiform rain, together with their total amount of precipitation and their occurrence frequency. As the first step toward the above purpose, the present study made a lesson practice for the university students by utilizing the 10-minute precipitation data for the four heavy rainfall events, in which the types of the heavy rainfall (although all the cases examined in the lesson are relating to the deep convective clouds) are rather different from each other, such as the differences of the rainfall intensity at the peak time, short-period variation of the rainfall intensity and the persistency of the rainfall including the "not so intense rainfall". The reports by the students seem to perceive the different features among these events briefly, but the students' attention to how long the intense rainfall with short-period variation or "not so intense rainfall" lasted was not so sufficient.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841762025Severe Anemia Caused by a Colorectal Lipoma With Central Erosions: A Case Reporte85768ENYusukeYoshidaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesRyoheiShojiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYukiMatsumiDepartment of Gastroenterological Surgery, Okayama University HospitalKoWatanabeDepartment of Pathology, Okayama University HospitalToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesColorectal lipomas are benign tumors that are often asymptomatic and discovered incidentally. In most cases, they can be managed conservatively with observation. We report the case of a man in his 70s with a colorectal lipoma located in the cecum. An investigation into his severe anemia led to the suspicion that the cecal lipoma was the underlying cause. An ileocecal resection was performed. Erosions were observed at the center of the lipoma. Although small colorectal lipomas are generally asymptomatic and rarely cause anemia, periodic endoscopic examinations are recommended. These lesions should be considered in the differential diagnosis of lower gastrointestinal bleeding.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1364-67532712026Compound heterozygosity of a novel missense variant and exonic deletion in hypomyelinating leukodystrophy 1516ENAkihikoMitsutakeDepartment of Neurology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan Department of NeurologyTakashiMatsukawaDepartment of Neurology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan Department of NeurologyKentaOrimoDepartment of Neurology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan Department of NeurologyKunihiroUedaDepartment of Neurology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan Department of NeurologyTomonariSekiDepartment of Neurology, Tokyo Teishin HospitalYasushiShiioDepartment of Neurology, Tokyo Teishin HospitalJunMitsuiDepartment of Precision Medicine Neurology, Graduate School of Medicine, The University of TokyoHiroyukiIshiuraDepartment of Neurology, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineHarushiMoriDepartment of Radiology, School of Medicine, Jichi Medical University,ShojiTsujiInstitute of Medical Genomics, International University of Health and WelfareTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan Department of NeurologyHypomyelinating leukodystrophy 15 (HLD15) results from biallelic pathogenic variants in EPRS1, but exonic deletions have not been reported. We describe a 40-year-old woman with mild intellectual disability, ataxia, dystonia, and MRI showing hypomyelination. Whole-exome sequencing identified a heterozygous missense variant in the prolyl-tRNA synthetase domain of EPRS1 (c.3430 C > G; p.Leu1144Val, NM_004446.3), without second variant. Whole-genome sequencing revealed a heterozygous 220-bp deletion spanning exon 15 (c.1743-30_1932del), and segregation analysis confirmed compound heterozygosity. RT-PCR from lymphoblastoid cells demonstrated exon-15 skipping leading to a frameshift (p.Asn582Serfs*10) and nonsense-mediated decay, leaving predominant expression of the paternally inherited missense allele. These findings support loss-of-function for the deletion and classify c.3430 C > G as likely pathogenic under ACMG/AMP criteria (PM1, PM2, PM3, PP3). This case represents the first exonic deletion reported in EPRS1. The relatively mild, adult-onset phenotype broadens both mutational and clinical spectra of HLD15 and highlights the importance of structural-variant analNo potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841772025The Challenge of Diagnosing Scirrhous Gastric Cancer by Endoscopic Biopsy: A Case Reporte87334ENYukaIkedaDepartment of Internal Medicine, Clinic IkedaDepartment of Internal Medicine, Clinic IkedaMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTadashiYoshinoDepartment of Pathology, Okayama UniversityTakehiroTanakaDepartment of Pathology, Okayama University HospitalNobumasaIkedaDepartment of Internal Medicine, Clinic IkedaMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesScirrhous gastric cancer, also known as linitis plastica, is a rare and aggressive subtype of gastric carcinoma that poses significant diagnostic challenges due to its submucosal infiltration and often normal-appearing mucosa. We report a case involving a 30-year-old Japanese woman who presented with a six-month history of epigastric pain and postprandial vomiting. Initial endoscopic examination revealed erythema and mucosal swelling, with limited antral distensibility and resistance during duodenal intubation. Despite 12 mucosal biopsies, histopathological examination revealed no evidence of malignancy. Given the strong clinical and endoscopic suspicion of scirrhous gastric cancer, additional deep sections and immunohistochemical staining were performed. These revealed scattered signet-ring cell carcinoma and poorly differentiated adenocarcinoma, with positive immunostaining for p53 and Ki67. The patient underwent total gastrectomy, and the diagnosis of scirrhous gastric cancer was confirmed on the resected specimen. This case highlights the importance of a high index of clinical suspicion, close collaboration between endoscopists and pathologists, and the utility of ancillary diagnostic tools, such as immunohistochemistry, in identifying subepithelial gastric malignancies that may be missed on conventional biopsy.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2577-171X922026Mitrofanoff Appendicovesicostomy With Boari Flap for Complete Female Urethral Transection: A Case Reporte70154ENKoheiMoriDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakehiroIwataDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTatsushiKawadaDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakuyaSadahiraDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYusukeTominagaDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSatoshiKatayamaDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShingoNishimuraDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKensukeBekkuDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYuichiroYamasakiDepartment of Urology, Kanagawa Children's Medical CenterMotooArakiDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesIntroduction: Female urethral complete transection caused by pelvic trauma is extremely rare, and no standard management has been established when urethral reconstruction is not feasible.<br>
Case Presentation: A woman in her twenties sustained an open pelvic fracture with perineal injury due to a traffic accident. Complete urethral transection was identified, and a suprapubic cystostomy was placed. After staged vaginal reconstruction and bladder function evaluation, a Mitrofanoff appendicovesicostomy was performed. Because the appendix was not enough to reach the umbilicus, a Boari flap was created to compensate for the length. Urodynamic evaluation showed improvement from a preoperative high-pressure bladder to increased compliance postoperatively, though pharmacological management was still required. Postoperatively, the patient achieved stable clean intermittent catheterization without complications.<br>
Conclusion: The Mitrofanoff procedure can be an effective option in female urethral injuries where reconstruction is impossible. The addition of a Boari flap may expand its applicability by overcoming conduit length limitations.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2590-1397282025Flow diverter treatment for internal carotid artery aneurysm following management of distal cerebral aneurysms: Technical note100540ENYuichiHirataDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasafumiHiramatsuDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenjiSugiuDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFukikoBabaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJuntaroFujitaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYutaSotomeDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasatoKawakamiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyuKimuraDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiEbisudaniDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunHarumaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomohitoHishikawaDepartment of Neurosurgery, Kawasaki Medical SchoolShotaTanakaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesBackground: In recent years, the effectiveness of flow diverters (FDs) for the treatment of intracranial aneurysms has been reported. While FDs are effective, their deployment involves advancing a delivery wire distally, which may pose a risk if a distal aneurysm exists within the same artery. In such cases, the delivery wire could potentially perforate the distal aneurysm. Here, we present two cases of tandem aneurysms in which an internal carotid artery (ICA) aneurysm was treated with an FD following the treatment of a distal cerebral aneurysm.<br>
Case description: A 44-year-old woman and a 67-year-old woman underwent magnetic resonance imaging for headache or abducens nerve palsy. In both cases, two aneurysms were revealed: one at the ICA and the other either at the middle cerebral artery or the top of the ICA. Due to the risk of perforation by the delivery wire during FD deployment, the distal aneurysms were treated first―either with surgical neck clipping or stent-assisted coil embolization. One month after the initial treatment, FD placement for the ICA aneurysm was performed as planned without complications in either case.<br>
Discussion: This is the first report where tandem aneurysms were successfully treated with treatment for distal cerebral aneurysms, followed by FDs for proximal ICA aneurysms. We emphasize the potential risk of perforation of the distal aneurysm by the delivery wire during FD placement.<br>
Conclusion: Treatment of distal cerebral aneurysms beforehand can help ensure the safe and effective use of FDs in patients with tandem aneurysms.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-818417112025A Case of Psoas Abscess Diagnosed With Oral Bacteria as the Causative Pathogene97584ENKokiUmemoriOral and Maxillofacial Surgery, Okayama University Graduate School of MedicineKyoichiObataOral and Maxillofacial Surgery, Okayama University Graduate School of MedicineMayumiYaoDentistry and Dental Surgery, Tsuyama Central HospitalKojiFujitaGeneral Internal Medicine and Infectious Diseases, Tsuyama Central HospitalSoichiroIbaragiOral and Maxillofacial Surgery, Okayama University Graduate School of MedicineWe report a rare case of a psoas abscess in an 87-year-old woman, in which oral commensal bacteria may have disseminated hematogenously from a chronic oral infection site and served as the causative pathogens. The patient presented with persistent left buttock pain, fever, and swelling, and imaging revealed a fracture of the left iliac bone with an associated psoas abscess. Bacterial cultures identified Streptococcus oralis and Pseudomonas aeruginosa. Her symptoms improved following antibiotic therapy and CT-guided drainage. Although the presence of P. aeruginosa in the oral cavity is generally considered transient, it has been isolated from the oral cavities of elderly and immunocompromised individuals. In the absence of lacerations or other direct portals of entry, and considering the identification of both pathogens, the oral cavity was regarded as the most likely source of infection. This case highlights the importance of correlating culture results with the most probable source of infection to improve the prognosis of systemic infections.No potential conflict of interest relevant to this article was reported.Ovid Technologies (Wolters Kluwer Health)Acta Medica Okayama2210-26121332025Robotic pancreatoduodenectomy for a giant duodenal leiomyoma: A case report and literature review111546ENSusumuDoitaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKoseiTakagiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMotohikoYamadaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuyaYasuiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomokazuFujiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesIntroduction: Duodenal leiomyomas are rare mesenchymal tumors. To date, several studies have reported on the safety and feasibility of surgical intervention for duodenal leiomyomas. However, minimally invasive surgery has rarely been performed in cases with duodenal leiomyomas. Herein, we present a case of a giant duodenal leiomyoma successfully treated with robotic pancreatoduodenectomy (RPD).<br>
Presentation of case: A 74-year-old man was referred to our hospital with a 6.5 cm duodenal tumor accompanied by gastrointestinal bleeding. The tumor was located in the second portion of the duodenum. Considering the tumor size and location, RPD was performed. Using the mesenteric Kocker maneuver, the posterior side of the duodenum was safely dissected, and the tumor was resected. The operative time was 373 min, with an estimated blood loss of 10 mL. The patient was followed up for 7 months with no recurrence.<br>
Discussion: To the best of our knowledge, this is the first to highlight the clinicopathological findings of a patient with duodenal leiomyoma undergoing RPD. To date, there have been 19 cases, including our case, reporting surgically treated duodenal leiomyoma. Treatment strategies should be decided depending on tumor characteristics, including the size, location, and histology of the tumor.<br>
Conclusion: We present a rare case of a giant duodenal leiomyoma that was successfully treated with RPD. Minimally invasive surgery can be safe and an alternative for the treatment of large duodenal tumors.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama1758-59021812025Metachronous Pancreatic Metastasis of Myxoid Liposarcoma Successfully Treated With Robotic Spleen‐Preserving Distal Pancreatectomy With Splenic Vessels Resections: A Case Reporte70069ENYumiSotaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKoseiTakagiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMotohikoYamadaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomokazuFujiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuyaYasuiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTakeyoshiNishiyamaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYasuoNagaiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesNoriyukiKanehiraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesAkariMasunagaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesPancreatic metastasis of myxoid liposarcoma (MLS) after primary resection is extremely rare. Herein, we present a case of metachronous pancreatic metastasis of MLS that was successfully treated with robotic spleen-preserving distal pancreatectomy (SPDP) using the Warshaw technique. A 60-year-old woman underwent radical resection of a 25-cm MLS in the right thigh after receiving neoadjuvant radiotherapy. The patient developed a 6-cm solitary pancreatic metastasis of the MLS 2 years later. Because no other distant metastases were detected, robotic SPDP (Warshaw technique) was performed. The operative time was 140 min with minimal blood loss. Follow-up at 3 months showed no recurrence. To our knowledge, this is the first report of a case of metachronous pancreatic metastasis of MLS successfully treated with robotic SPDP. Curative resection using minimally invasive surgery should be performed for solitary pancreatic metastases from MLS.No potential conflict of interest relevant to this article was reported.Japanese Society of Internal MedicineActa Medica Okayama0918-291864142025Myeloid Sarcoma in the Small Intestine21552159ENMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomohiroKamioDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesShoichiroHirataDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKatsunoriMatsuedaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesDaisukeKametakaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTakehiroTanakaDepartment of Pathology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesSeijiKawanoDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMyeloid sarcoma is a rare extramedullary tumor of immature myeloid cells that is often associated with acute myeloid leukemia (AML). We herein report an 81-year-old man who presented with intestinal obstruction due to myeloid sarcoma of the small intestine. Diagnostic challenges were overcome using double-balloon enteroscopy and a biopsy, which confirmed the diagnosis of myeloid sarcoma. The patient subsequently developed AML but responded well to chemotherapy. This case underscores the importance of considering myeloid sarcoma in the differential diagnosis of small-bowel tumors. Highlighting the significance of a histological analysis, even in patients presenting with small bowel obstruction, the early diagnosis and treatment are crucial for improving outcomes, particularly in patients without a history of hematologic malignancies.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841752025Pseudoachalasia Due to Malignant Pleural Mesothelioma Involving the Esophaguse84161ENManamiHondaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuyaMiyamotoDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTakehiroTanakaDepartment of Pathology, Okayama University HospitalMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesWe report a rare case of pseudoachalasia secondary to malignant pleural mesothelioma involving the esophagus. A 66-year-old man presented with progressive dysphagia, weight loss, and postprandial hiccups. Endoscopic examination showed esophageal dilation with luminal narrowing at the esophagogastric junction, but no mucosal abnormalities. Computed tomography revealed an irregular-shaped mass extending from the peri-esophagogastric junction to the retroperitoneum, accompanied by pleural effusion, right-sided hydronephrosis, and multiple hepatic lesions. Endoscopic ultrasound-guided fine-needle aspiration from the mass lesion through the esophageal lumen revealed epithelioid malignant mesothelioma. This case highlights the importance of considering malignant mesothelioma in the differential diagnosis of pseudoachalasia, particularly when imaging reveals extrinsic esophageal compression without mucosal lesions.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841742025Gastrointestinal Stromal Tumors in the Stomach With Tumor Growth and Hemorrhage During Conservative Management: A Report of Two Casese82046ENMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesSatoruKikuchiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesShinjiKurodaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTakehiroTanakaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesGastrointestinal stromal tumors (GISTs) are often detected incidentally during esophagogastroduodenoscopy. Although surgical resection is the standard treatment for GISTs, patients with significant comorbidities may not be eligible for surgery and are managed conservatively. Herein, we report two cases of gastric GISTs that were initially observed during the management of other comorbidities but subsequently became enlarged, resulting in gastrointestinal bleeding. These cases highlight the potential risks of tumor progression and bleeding in patients undergoing conservative management.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841722025Gastric Metastasis of Renal Cell Carcinoma Initially Diagnosed by Esophagogastroduodenoscopye79651ENMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomohiroKamioDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesShoichiroHirataDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTakehiroTanakaDepartment of Pathology, Okayama University HospitalMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesHere, we report a rare case of renal cell carcinoma (RCC) initially detected as a gastric metastasis. A 58-year-old man with epigastric discomfort underwent esophagogastroduodenoscopy, which revealed a reddish semi-pedunculated lesion with a whitish coating. Biopsy and imaging confirmed clear cell RCC metastasis. Contrast-enhanced computed tomography (CT) revealed a primary renal tumor with pancreatic and lymph node metastases. Despite chemotherapy treatment, the patient died after 10 months. Gastric metastases from RCC, although rare, should be considered in highly vascular gastric lesions with white coatings. Clinicians must be vigilant for metastatic diseases with atypical gastric findings.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2950-2217322025Erythromelalgia presenting with posterior reversible encephalopathy syndrome: A pediatric case report100078ENKengoSuzukiDepartment of Pediatrics, Okayama University HospitalKazuhiroUdaDepartment of Pediatrics, Okayama University HospitalMitsuruTsugeDepartment of Pediatric Acute Diseases, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKyosukeArakawaDepartment of Anesthesiology and Resuscitology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenjiShigeharaDepartment of Pediatrics, Okayama University HospitalTakafumiObaraDepartment of Emergency, Critical Care, and Disaster Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKoseiHasegawaDepartment of Pediatrics, Okayama University HospitalHirokazuTsukaharaDepartment of Pediatrics, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesBackground: Erythromelalgia is a rare disorder characterized by erythema, warmth, and burning pain in the extremities. We report a pediatric case of erythromelalgia in a patient who developed posterior reversible encephalopathy syndrome (PRES), without any cutaneous signs.<br>
Case presentation: A previously healthy 12-year-old girl presented to our pediatric clinic with burning extremity pain that had persisted for 6 weeks. The patient was treated with analgesics; however, the pain was refractory to these agents. Seven days after the first visit, she developed afebrile seizures and was transferred to our hospital. Her initial blood pressure was 139/105 mmHg (+2.0 SD), and brain magnetic resonance imaging revealed high intensity areas in the bilateral parietal and occipital lobes, leading to a diagnosis of PRES. Her blood pressure was difficult to control with anti-hypertensive agents. Burning pain in her extremities was relieved by cooling and worsened by warming. Although erythema was not observed in her hands or legs, erythromelalgia was suspected based on the characteristic nature of her pain. Intravenous lidocaine was administered for diagnosis, which was dramatically effective. After initiating mexiletine, the burning pain in her extremities disappeared, and hypertension improved. A final diagnosis of erythromelalgia with PRES was made.<br>
Conclusion: A history of temperature-dependent pain relief and deterioration are important indicators of disease diagnosis, even if patients indicate a lack of erythema or warmth. Physicians should be aware that persistent pain due to erythromelalgia can lead to refractory hypertension and development of PRES.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-09041422026Oculogyric Crisis During Chronic Aripiprazole Therapy: A Diagnostic Challenge in the Emergency Departmente72067ENKoheiTokiokaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama Universitye, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences Okayama University JapanTsuyoshiNojimaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityIppeiMatsuoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityKoheiTsukaharaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityAtsunoriNakaoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityOculogyric crisis can occur even during chronic, stable aripiprazole therapy without recent dose escalation. In patients with acute upward eye deviation and an otherwise normal neurologic examination, medication review is key to recognizing drug-induced dystonia and avoiding unnecessary neurologic workup.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-818417122025Streptococcal Toxic Shock Syndrome Following Intestinal Obstruction in a Patient With Crohn’s Diseasee100138ENAyanoNishioDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University HospitalToshihiroInokuchiDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMikakoIshiguroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYukiAoyamaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMasahiroTakaharaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesSakikoHiraokaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKoheiOguniDepartment of Infectious Diseases, Okayama University HospitalHideharuHagiyaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYutoMatsuokaDepartment of Anesthesiology and Resuscitology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomoyukiKanazawaDepartment of Anesthesiology and Resuscitology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesStreptococcal toxic shock syndrome (STSS) is a rare, life-threatening complication of invasive group A streptococcal (iGAS) infections. We report the case of a 24-year-old woman with Crohn's disease receiving immunosuppressive therapy who developed STSS following intestinal obstruction. On day 2, she developed fever, altered mental status, hypoxemia, erythema, and hypotension. Chest CT revealed bilateral pulmonary infiltrates, and blood cultures grew emm1-positive M1UK Streptococcus pyogenes, confirming STSS. Early multidisciplinary intervention resulted in rapid recovery without sequelae. This case emphasizes the importance of considering iGAS-induced STSS in septic shock, especially in immunocompromised patients, and highlights the need for prompt recognition and treatment.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X8012026Effective Treatment of Advanced Hepatocellular Carcinoma with Extensive Peritoneal Dissemination Using Lenvatinib6974ENShinyaWakatsukiDepartment of Obstetrics and Gynecology, Kochi Health Sciences CenterShinyaSakamotoDepartment of Gastroenterological Surgery, Kochi Health Sciences CenterAkikoUenoDepartment of Obstetrics and Gynecology, Kochi Health Sciences CenterTakaomiNambaDepartment of Obstetrics and Gynecology, Kochi Health Sciences CenterYoritoYamamotoDepartment of Obstetrics and Gynecology, Kochi Health Sciences CenterManabuMatsumotoDepartment of Diagnostic Pathology, Kochi Health Sciences CenterJunIwataDepartment of Diagnostic Pathology, Kochi Health Sciences CenterTakehiroOkabayashiDepartment of Gastroenterological Surgery, Kochi Health Sciences CenterCase Report10.18926/AMO/70075Patients with hepatocellular carcinoma (HCC) and extensive peritoneal dissemination generally have a poor prognosis and are often resistant to systemic therapy. We report the case of a 47-year-old woman with HCC and massive peritoneal dissemination who presented with malignant ascites requiring repeated cell-free and concentrated ascites reinfusion therapy and peritoneovenous shunt placement, as well as malignant pleural effusion requiring pleurodesis. Combined immunotherapy with durvalumab/tremelimumab was initiated;however, disease progression was observed after three treatment courses, prompting a switch to lenvatinib therapy. Two months after initiation of lenvatinib, CT imaging demonstrated complete disappearance of arterial enhancement in the primary hepatic lesion, along with reduction in the size of peritoneal dissemination nodules. Thirteen months after switching to lenvatinib (16 months after the initial diagnosis), the alpha-fetoprotein level continued to decrease, and the disease remained stable under treatment. Despite the extremely high tumor burden, lenvatinib achieved disease stabilization and symptomatic improvement.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X8012026Metastatic Intraocular Tumor Likely from Hepatocellular Carcinoma Mimicking Panuveitis6367ENEriTakasuDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYusukeShiodeDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHiroyaKindoDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShuheiKimuraDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMioHosokawaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyoMatobaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiKanzakiDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTetsuroMoritaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakuyaAdachiDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiMorizaneDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/70074A 77-year-old man undergoing treatment for hepatocellular carcinoma (HCC) presented with blurred vision in his right eye, persisting for 2 months. Slit-lamp microscopy and fundus examination revealed inflammatory cells in the anterior chamber, severe vitreous opacities, and retinal vasculitis in the right eye. The patient underwent vitreous surgery with biopsy, and vitreous cytology confirmed a metastatic intraocular tumor originating from the HCC. Radiotherapy was administered to the right eye, with no recurrence of intraocular inflammation observed at 10 months post-irradiation.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2769-2558512026Cardiogenic cerebral infarction after Takotsubo cardiomyopathy in a patient with catatonia: A case reporte70285ENMasakiFujiwaraDepartment of Neuropsychiatry, Medical Development Field, Okayama UniversityYutoYamadaDepartment of Neuropsychiatry, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuhiroOsawaDepartment of General Internal Medicine 3, Kawasaki Medical School General Medical CenterShinjiSakamotoDepartment of Neuropsychiatry, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesMasafumiKodamaOkayama Psychiatric Medical CenterBackground: Takotsubo cardiomyopathy (TTC) is a transient cardiac condition often triggered by an emotional or physical stress. TTC usually has a benign clinical course with full recovery. However, in rare cases, TTC is complicated by cardiogenic shock, left ventricular rupture, or ventricular thrombus. We report a case of a patient with catatonia who developed TTC and subsequently experienced extensive cerebral infarction.<br>
Case Presentation: A 71-year-old woman with no prior psychiatric history was admitted for catatonia following a suicide attempt. During hospitalization, she exhibited electrocardiography (ECG) abnormalities and elevated D-dimer levels. Transthoracic echocardiography revealed apical hypokinesis and basal hyperkinesis, consistent with TTC, along with an intraventricular thrombus. Cardiovascular CT angiography confirmed normal coronary arteries. She was diagnosed with TTC complicated by left ventricular thrombus and deep vein thrombosis. Anticoagulant therapy was initiated. Despite improvement in catatonia with lorazepam, she developed right hemiplegia and aphasia on Day 5 due to cardiogenic cerebral infarction from thromboembolism. Thrombolytic therapy was not indicated, and conservative treatment was provided. Although cardiac function normalized by Day 16, she was left with severe neurological deficits.<br>
Conclusion: The case highlights the diagnostic challenges of TTC in non-communicative psychiatric patients and the potential for severe complications. Psychiatrists need to be aware of the development of TTC as a serious physical complication in patients with catatonia.No potential conflict of interest relevant to this article was reported.SAGE PublicationsActa Medica Okayama1756-2848192026Clinical efficacy and safety of endoscopic ultrasound-guided ablation therapies for pancreatic neuroendocrine tumors: a systematic review and meta-analysisENKazuyukiMatsumotoDepartment of Gastroenterology and Hepatology, Okayama University HospitalYukiFujiiDepartment of Gastroenterology and Hepatology, Okayama University HospitalDaisukeUchidaDepartment of Gastroenterology and Hepatology, Okayama University HospitalYasutoTakeuchiDepartment of Gastroenterology and Hepatology, Okayama University HospitalToshiharuMitsuhashiCenter for Innovative Clinical Medicine, Okayama University HospitalMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University HospitalBackground: Pancreatic neuroendocrine tumors (pNETs) are rare; however, they are increasingly being detected. Although surgical resection remains the standard treatment, its invasiveness has prompted interest in less invasive alternatives, particularly for small non-functional pNETs (NF-pNETs) and insulinomas.<br>
Objectives: To evaluate the clinical efficacy and safety of endoscopic ultrasound-guided ethanol injection (EUS-EI) and radiofrequency ablation (EUS-RFA) for pNETs.<br>
Design: A systematic review and meta-analysis.<br>
Data sources and methods: A literature search of PubMed, MEDLINE, and Google Scholar was conducted (April 2005–April 2025). Studies were eligible if they reported clinical outcomes of EUS-EI or EUS-RFA in adult patients with insulinomas or NF-pNETs. The primary endpoints were clinical success (short-term symptom resolution or radiological response) and adverse event (AE) rates. Data were pooled using a random-effects model.<br>
Results: Twenty-six studies were included in the meta-analysis. For insulinomas, the pooled clinical success rate was 77% (95% confidence interval (CI), 59–88) for EUS-EI and 95% (95% CI, 89–97) for EUS-RFA. The pooled incidence of total AEs was 32% (95% CI, 17–51) for EUS-EI and 25% (95% CI, 15–39) for EUS-RFA. For NF-pNETs, the pooled clinical success rates were 76% (95% CI, 54–90) for EUS-EI and 85% (95% CI, 74–92) for EUS-RFA, and the pooled incidence of total AEs was 27% (95% CI, 20–35) and 26% (95% CI, 17–38), respectively. The most common moderate or severe AEs were pancreatitis in 12 patients (7.6%) after EUS-EI, and pancreatic fluid collection in 4 patients (1.9%) and pancreatic duct stricture in 3 patients (1.4%) after EUS-RFA. One fatal case occurred in a 97-year-old patient following EUS-RFA.<br>
Conclusion: Both EUS-EI and EUS-RFA are effective, relatively safe, and minimally invasive treatment options for pNETs. However, severe AE can occur, and careful patient selection and treatment indication are essential.<br>
Trial registration: Not registered.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1349-41475412026Mycobacterium mageritense-associated refractory cutaneous infection and lymphadenitis in an immunocompetent adult: insights from genomic sequencing19ENShinnosukeFukushimaDepartment of Bacteriology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineJumpeiUchiyamaDepartment of Bacteriology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYoshioKawakamiDepartment of Dermatology, Okayama University HospitalYoshikoMatsuuraKonohana Dermatology ClinicSatoruSugiharaDepartment of Dermatology, Okayama University HospitalShinMorizaneDepartment of Dermatology, Okayama University HospitalPoowadonMuenrayaDepartment of Bacteriology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalBackground Nontuberculous mycobacteria are increasingly recognized as causes of chronic and refractory skin and soft tissue infections, even in individuals without immunodeficiency. Among them, Mycobacterium mageritense is a rare, rapidly growing species that can lead to persistent lesions requiring prolonged antimicrobial therapy. Reports of M. mageritense infections involving both the skin and regional lymph nodes are limited, and this case adds new clinical and genomic insights.<br>
Case presentation A 48-year-old previously healthy man presented with a slowly enlarging cutaneous lesion on his lower leg and ipsilateral inguinal lymphadenitis. Empirical antibacterial therapy with β-lactams and macrolides was ineffective. Wound cultures subsequently grew M. mageritense, confirmed by whole-genome sequencing. Several antimicrobial regimens were attempted, and the final successful therapy consisted of oral levofloxacin and minocycline for 4 months, leading to complete clinical resolution. Genomic analysis identified resistance-related genes, including erm(40), aac(2′)-Ib, tet(V), and RbpA, although in vitro minimum inhibitory concentrations showed variable susceptibility. Phylogenetic comparison revealed that the isolate was closely related to previously reported M. mageritense strains from Japan.<br>
Conclusions This case demonstrates that M. mageritense can cause cutaneous infection with secondary lymphadenitis in an immunocompetent host. Accurate species identification using molecular or genomic methods and selection of appropriate combination antibiotic therapy based on susceptibility testing are crucial for successful management of such infections.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841732025Prospective Evaluation of the Safety and Compression Performance of Novel Compression Denim Jeans in Healthy Volunteers and Patients With Lymphedemae80971ENDaikiOusakaDepartment of Pharmacology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKiyoshiYamadaDepartments of Plastic and Reconstructive Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesNorikoSakanoDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSatoeKirinoDepartment of Obstetrics and Gynecology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazumasaMiyakeDepartment of Rehabilitation, Lymphedema Treatment Center, Kousei HospitalTakumiTakahashiDivision of Business Management, Matsuoka CorporationAkihiroMatsuokaDivision of Production Engineering, Matsuoka CorporationShintaroYamadaDivision of Sales, Kaihara CorporationAkiraShinaokaDepartment of Lymphatics and Edematology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSusumuOozawaDepartment of Clinical Safety, Okayama University HospitalObjectives: The treatment of lower-extremity lymphedema, whether congenital or acquired, remains challenging. Long-term management aimed at reducing complications and maximizing quality of life is essential. Compression stockings are crucial in this management; however, their application is limited by patient experience (ease of wear, texture, breathability, and appearance). This highlights the need to evaluate alternative compression garments that maintain therapeutic efficacy while improving patient adherence.<br>
Methods: We developed a novel compression denim product (Flow plus Jeans®) using advanced sewing technology. Its baseline performance (compression ability) was evaluated by measuring pressure gradients at three points (ankle, calf, and thigh) using a mannequin-based compression testing system and compared with those of existing stockings. Thereafter, a safety assessment was conducted on healthy volunteers to evaluate potential adverse effects, including changes in lower limb circumference, signs of deep vein thrombosis (DVT) via ultrasound, and skin complications. A clinical trial in patients with lymphedema was then performed to compare its efficacy with that of conventional compression stockings.<br>
Results: Baseline performance testing with a mannequin revealed that Flow plus Jeans demonstrated compression levels and pressure gradients at three calf points comparable to those of standard compression stockings. A safety study involving nine healthy volunteers confirmed that Flow plus Jeans caused no significant changes in lower-limb circumferences after three days of wear, with no cases of DVT or skin complications. In a subsequent clinical trial involving nine female patients with lymphedema, the jeans showed non-inferiority to existing stockings concerning lower-limb circumference measurements at six points (pre-use vs. six months post-use), with patient-reported experiences assessed via questionnaires. Notably, patients reported enhanced satisfaction regarding the jeans' fashionability, which could serve as an incentive for long-term adherence.<br>
Conclusion: Our findings suggest that Flow plus Jeans represent a promising novel option for the long-term management of lymphedema, offering an alternative that balances medical efficiency with improved patient satisfaction and demonstrates safety in healthy individuals.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841812026Central Serous Chorioretinopathy in Parallel With Onset and Relapses of Minimal Change Nephrotic Syndrome: A 28-Year Case Follow-Upe102426ENToshihikoMatsuoDepartment of Ophthalmology, Graduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityJunWadaDepartment of Nephrology, Rheumatology, Endocrinology, and Metabolism, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityCentral serous chorioretinopathy is an idiopathic disease that manifests as one or several localized, small, dome-shaped serous retinal detachments on fundus examination. The pathophysiology involves fluid leakage from the choroidal capillaries, known as the choriocapillaris, into the subretinal space through sites of damage in the retinal pigment epithelium. This case report discusses the underlying causes of central serous chorioretinopathy-like findings in minimal change nephrotic syndrome.<br>
<br>
The patient was a 33-year-old woman who developed nephrotic syndrome that was confirmed to be minimal change disease by renal biopsy. She experienced two major relapses of nephrotic syndrome at the ages of 36 and 41 years. She also had a minor relapse at the age of 37 years, five months after the first major relapse at the age of 36 years, as well as four additional minor relapses at the ages of 44, 46, 50, and 51 years. The onset of central serous chorioretinopathy-like manifestations, which were localized to the left eye, occurred three months after the initial onset of nephrotic syndrome at the age of 33 years. Two subsequent episodes of relapse of central serous chorioretinopathy-like manifestations were observed in both eyes at intervals of five months and one month, respectively, after major relapses of nephrotic syndrome at the ages of 36 and 41 years. Thereafter, she did not develop further central serous chorioretinopathy-like manifestations.<br>
<br>
She discontinued oral prednisolone at the age of 54 years and experienced no further relapses of nephrotic syndrome through her latest visit at the age of 61 years. She maintained normal renal function and good visual acuity in both eyes. The long-term, consistent temporal association between episodes of central serous chorioretinopathy and the onset and relapses of minimal change nephrotic syndrome is strongly supported by longitudinal clinical observations spanning 28 years. This parallel course suggests a possible shared pathophysiological mechanism or common triggering factors underlying both diseases.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841812026Endoscopic Topical Application (ETA) Therapy for Refractory Overactive Bladder: A First-in-Human Reporte101143ENTakuyaSadahiraDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasahiroSugiharaDepartment of Surgery, Nishi Fukuyama HospitalYosukeMitsuiDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesToyohikoWatanabeDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMotooArakiDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasamiWatanabeDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRefractory overactive bladder (OAB) remains a clinical challenge despite established therapies, such as anticholinergics, β3-agonists, and intradetrusor botulinum toxin (BTX). Emerging evidence suggests that sensory mechanisms within the bladder, including those involving the trigone where superficial afferent networks are present, may contribute to persistent urinary urgency and frequency in some patients. Although intradetrusor BTX injection is effective in selected patients, its impact on these superficial pathways may be limited because the injected drug predominantly distributes within the detrusor. Endoscopic topical application (ETA) therapy delivers BTX directly to the trigone under air cystoscopy, potentially providing targeted modulation of sensory hyperexcitability. We report a 72-year-old woman with long-standing refractory OAB who experienced only partial improvement with repeated intradetrusor BTX injections but achieved clinically meaningful symptom relief after ETA therapy. Nocturia, urgency, urgency urinary incontinence, and voided volume were improved, with no complications other than transient postoperative urethral pain. This case suggests that ETA therapy may represent a promising sensory-focused option for refractory OAB.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama1341-321X31122025Whole-genome sequencing and in vitro characterization of a disseminated ST398 Staphylococcus aureus infection: A case report102845ENYosukeSazumiDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinnosukeFukushimaDepartment of Bacteriology, Okayama University, Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalAtsushiKatoDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAtsuhitoSuyamaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKoheiOguniDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazuyoshiGotohDepartment of Medical Laboratory Science, Okayama University Graduate School of Health SciencesShokoKutsunoAntimicrobial Resistance Research Center, National Institute of Infectious Diseases, Japan Institute for Health SecurityJunzoHisatsuneAntimicrobial Resistance Research Center, National Institute of Infectious Diseases, Japan Institute for Health SecurityMotoyukiSugaiAntimicrobial Resistance Research Center, National Institute of Infectious Diseases, Japan Institute for Health SecurityShumaTsujiDepartment of Bacteriology, Okayama University, Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKojiIioMicrobiology Division, Clinical Laboratory, Okayama University HospitalFumioOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesStaphylococcus aureus potentially causes systemic infections such as disseminated abscesses and bloodstream infections, leading to high mortality rates. We herein describe a case of disseminated muscle abscesses caused by sequence type (ST) 398 methicillin-sensitive S. aureus (MSSA), along with in vitro investigation results for potential pathogenic factors. A 67-year-old healthy woman was admitted to our hospital with complaints of systemic body pain. Blood cultures identified MSSA and contrast-enhanced computed tomography revealed multiple muscle abscesses extending from her neck to her soles. She received antibiotic treatment with intravenous cephazolin and underwent repeated surgical drainage, and was finally discharged. Notably, the MSSA strain exclusively affected her muscle tissues, prompting us to perform genetic analysis to uncover the underlying reason. Short-read genome analysis revealed the isolate to be ST398, harboring chp and scn genes known for immune evasion from human immunity. However, no other known pathogenic factors were identified despite rigorous assays for biofilm formation, surface and cell wall proteins, protease production, and hyaluronidase activity. ST398 S. aureus is commonly isolated from livestock, and her prior experience of being flooded could be related to the disease onset. The present case underscores the possibility of severe ST398 MSSA infections in humans, even in the absence of direct animal exposure.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-818417112025Perioperative Multidisciplinary Intervention Led to Complete Minimally Invasive Transthoracic Esophagectomy for a Patient With Severe Lung Dysfunction: A Case Reporte97931ENMakotoMatsumotoDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMasashiHashimotoDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKentoKawasakiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomoyoshiKunitomoDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesNaoakiMaedaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesShunsukeTanabeDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuhiroNomaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesRisk factors for postoperative pneumonia after esophagectomy include smoking, severe lung dysfunction, and sarcopenia. Heavy smokers often have chronic obstructive pulmonary disease (COPD), which is associated with poor physical activity and low muscle strength. Sarcopenia is also associated with decreased physical function and malnutrition. These factors lead to a close relationship between COPD and sarcopenia. This report describes the case of a 74-year-old man who presented with dysphagia and was diagnosed with advanced esophageal cancer with lymph node metastasis. Preoperative respiratory function testing showed a forced expiratory volume in one second (FEV1) of 0.76 L because of his past smoking and COPD. Multidisciplinary intervention was started, along with neoadjuvant chemotherapy. Preoperative management improved his physical function. Robot-assisted thoracoscopic subtotal esophagectomy with the patient in the prone position was performed with curative resection and no severe postoperative complications. The perioperative multidisciplinary intervention improved physical functions and enabled safe robot-assisted thoracoscopic esophagectomy for the patient with severe lung dysfunction in the prone position. This case highlights that not only respiratory status but also physical parameters should be taken into account when considering whether a patient can tolerate surgery safely.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama1758-59021812025A Procedural Transhiatal Approach for the Thoracic Para‐Aortic Lymph Node: A Case Reporte70066ENMasashiHashimotoDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazuhiroNomaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYasushigeTakedaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHijiriMatsumotoDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKentoKawasakiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomoyoshiKunitomoDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesNaoakiMaedaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShunsukeTanabeDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesThe thoracic posterior para-aortic lymph node (TPAN) is classified as an extra-regional lymph node in esophageal cancer, with metastasis indicating poor prognosis. However, some cases with suspected TPAN metastasis may benefit from esophagectomy with lymph node dissection, including TPAN. This report presents the case of a 58-year-old man with upper thoracic esophageal squamous cell carcinoma and suspected simultaneous TPAN metastasis who underwent neoadjuvant chemotherapy followed by thoracoscopic subtotal esophagectomy and procedural transhiatal TPAN dissection. This transhiatal approach provided direct access to the lymph node without additional thoracic incisions, ensuring safe resection in coordination with the assistant and following anatomical landmarks systematically. Pathological examination showed a false-positive TPAN finding, though the patient later developed distant recurrence. Compared with conventional approaches, this transhiatal technique allows for procedural and reproducible lymphadenectomy while minimizing respiratory burden. This case highlights the feasibility of a transhiatal approach for TPAN dissection.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-090413122025Experience and Insight Into Genetic Diagnosis of Infective Aortic Aneurysme71586ENShinnosukeFukushimaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHideharuHagiyaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakumiFujimoriMicrobiology Division, Clinical Laboratory, Okayama University HospitalKojiIioMicrobiology Division, Clinical Laboratory, Okayama University HospitalCulture-negative infected aneurysms possibly occur in various clinical situations, including prior antibiotic exposure. Accurate microbial identification is crucial for an optimal antimicrobial strategy. 16S ribosomal RNA gene sequence analysis would provide a useful tool for precise bacterial identification.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-090413122025Mycobacterium intracellulare-Induced Flexor Tenosynovitisof the Forearme71698ENHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalShinnosukeFukushimaDepartment of Infectious Diseases, Okayama University HospitalKentaNakamotoDepartment of Infectious Diseases, Okayama University HospitalKoheiOguniDepartment of Infectious Diseases, Okayama University HospitalWe herein document a clinically challenging case of Mycobacterium intracellulare tenosynovitis in an immunocompromised patient with rheumatoid arthritis. Diagnosing nontuberculous mycobacterial tenosynovitis is often challenging because it often mimics rheumatoid arthritis flares. Combination antimycobacterial therapy combined with surgical intervention is essential for the treatment of this refractory condition.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-090413102025Recurrent Septic Shock in Immunosuppressed Patientse71249ENShinnosukeFukushimaDepartment of Infectious Diseases, Okayama University HospitalKojiFujitaDepartment of General Medicine and Infectious Diseases, Tsuyama Chuo HospitalHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalCytomegalovirus gastroenteritis presents with diarrhea and abdominal pain in immunosuppressed patients, and histopathological examination is essential by endoscopy. This case illustrates that cytomegalovirus enteritis may develop insidiously and possibly invoke shock in immunocompromised patients, warranting its inclusion in the differential diagnosis of recurrent septic shock.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2051-33801362025Corynebacterium striatum-Associated Pyogenic Osteomyelitis With Direct Extension From Postoperative Empyemae70230ENShinnosukeFukushimaDepartment of Infectious Diseases, Okayama University HospitalKazuhiroNomaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalCorynebacterium striatum can cause postoperative empyema. C. striatum-associated empyema may be associated with osteomyelitis. Rifampicin is a viable therapeutic option for C. striatum infection.No potential conflict of interest relevant to this article was reported.Laser Institute of AmericaActa Medica Okayama1042-346X3712025High-quality welding of copper and aluminum by angled laser irradiation and insert metals012041ENYasuhiroOkamotoFaculty of Environmental, Life, Natural Science and Technology, Okayama UniversityYukiYamadaGraduate School of Environmental, Life, Natural Science and Technology, Okayama UniversityAkiraOkadaFaculty of Environmental, Life, Natural Science and Technology, Okayama UniversityMasakiMiyakeKataoka CorporationTakeshiYamamuraKataoka CorporationKatsutoshiNagasakiKataoka CorporationNorioNishiKataoka CorporationCopper is an important material in order to use electricity effectively, and aluminum is also an attractive material to perform lightweight construction. Laser welding of copper and aluminum is required, and there are some reports to weld copper and aluminum firmly by laser irradiation from the aluminum side. On the other hand, laser irradiation from the copper side requires high power density to generate a keyhole, which causes the strong convection of molten aluminum into copper. Thus, brittle intermetallic compounds with rich aluminum result in the deterioration of joining strength. Angled laser irradiation enables mild heat input to the aluminum side, and the combination of angled and superposed irradiation of blue and near-infrared (NIR) lasers achieved a remarkable increase of breaking strength. However, the breaking strength is still lower than the base materials. Therefore, insert materials between copper and aluminum plates were investigated by the angled irradiation of blue and NIR lasers from the copper side. The breaking strength with insert materials showed higher values in the order of titanium mesh, nickel foil, and nickel mesh, and its value with the nickel mesh was close to the tensile strength of aluminum in a cross tensile test. Further improvement of breaking strength is possible by inserting the nickel mesh between copper and aluminum plates in the case of angled irradiation of blue and NIR lasers even from the copper side.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-09041322025Maturity-Onset Diabetes of the Young (MODY) With HNF1B p.Glu105Lys Mutation Achieving Significant Insulin Reduction on Tirzepatide: A Case Reporte70173ENMihiroSueDepartment of Diabetology and Metabolism, NHO Okayama Medical Center MayuWatanabeDepartment of Diabetology and Metabolism, NHO Okayama Medical Center AyumiInoueDepartment of Diabetology and Metabolism, NHO Okayama Medical Center AkihiroKatayamaDepartment of Diabetology and Metabolism, NHO Okayama Medical Center SanaeTeshigawaraDepartment of Internal Medicine, Diabetes Center, Okayama Saiseikai General HospitalYuichiMatsushitaDepartment of Diabetology and Metabolism, NHO Okayama Medical Center MasayaTakedaDepartment of Diabetology and Metabolism, NHO Okayama Medical Center IzumiIsedaDepartment of Diabetology and Metabolism, NHO Okayama Medical Center JunEguchiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKazuyukiHidaDepartment of Diabetology and Metabolism, NHO Okayama Medical Center This report describes the first case of maturity-onset diabetes in young (MODY) with HNF1B mutation started administration of the dual glucose-dependent insulinotropic polypeptide and glucagon-like peptide-1 receptor agonist, tirzepatide. A 26-year-old female with a 15-year history of diabetes mellitus was diagnosed with MODY, which is characterized by decreased insulin secretion. She was treated with tirzepatide, which significantly improved her glycemic management; insulin secretion increased the fasting serum C-peptide immunoreactivity from 0.36 to 1.09 ng/mL. The patient discontinued glimepiride, and her total daily insulin dose was reduced from 88 to 4 units. This report highlights the glucose-lowering effects of tirzepatide in a patient with MODY who has the HNF1B p.Glu105Lys mutation.No potential conflict of interest relevant to this article was reported.The Editorial Committee of Annals of Vascular DiseasesActa Medica Okayama1881-641X1812025Successful Surgical Treatment of a Spontaneous Rupture of the Left Iliac Vein: What Is the Optimal and Radical Treatment?cr.25-00065ENKeiMoriokaDepartment of Cardiovascular Surgery, Okayama University HospitalMasanoriHirotaDepartment of Cardiovascular Surgery, Showa Medical University Fujigaoka HospitalShingoKasaharaDepartment of Cardiovascular Surgery, Okayama University HospitalSpontaneous rupture of the iliac vein (SRIV) requires surgical hemostasis and venous return restoration. We herein report a case treated with initial thrombus removal and direct venous repair. Because of early occlusion, a 2nd surgery was performed for iliac vein reconstruction using a 14-mm ringed Gore-Tex graft (W. L. Gore & Associates, Newark, DE, USA), and a 4-mm Gore-Tex arteriovenous shunt was created between the femoral artery and the femoral vein to prevent reocclusion. The patient had an uneventful recovery without recurrence. A single-stage procedure including hemostasis, vein replacement, and arteriovenous bypass may be ideal for radical SRIV treatment.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2467-981X102025Favorable outcomes of epilepsy with gait-induced seizures after resection of the unilateral supplementary motor area489492ENSatoshiKodamaDepartment of Neurology, Graduate School of Medicine, The University of TokyoNaotoKuniiDepartment of Neurosurgery, Jichi Medical UniversityYuichiroShirotaDepartment of Neurology, Graduate School of Medicine, The University of TokyoTakuseiChouDepartment of Neurology, Graduate School of Medicine, The University of TokyoMizuhoKawaiDepartment of Neurology, Graduate School of Medicine, The University of TokyoSeijiroShimadaDepartment of Neurosurgery, Graduate School of Medicine, The University of TokyoMeikoMaedaDepartment of Neurology, Graduate School of Medicine, The University of TokyoHiroyukiIshiuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasashiHamadaDepartment of Neurology, Graduate School of Medicine, The University of TokyoMasakoIkemuraDepartment of Pathology, Graduate School of Medicine, The University of TokyoYukoSaitoDepartment of Neuropahtology (Brain Bank for Aging Research), Tokyo Metropoliran Institute for Geriatrics and GerontologyNaokiAkamatsuDepartment of Neurology, International University of Health and Walfare Narita HospitalTairaUeharaDepartment of Neurology, International University of Health and Walfare Narita HospitalNobuhitoSaitoDepartment of Neurosurgery, Graduate School of Medicine, The University of TokyoTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of TokyoBackground: Gait-induced seizures are a rare manifestation of reflex epilepsy. Pathophysiology of this phenomenon has not been fully understood.<br>
Case presentation: A 28-year-old woman presented with a long history of “falls” following paroxysmal bilateral leg stiffness triggered by walking. Scalp electroencephalogram (EEG) revealed low-amplitude rhythmic beta activity, maximal at the Cz electrode, during these events. Magnetoencephalography demonstrated repetitive sharp waves source-localized to the right primary motor cortex. Multiple anti-seizure medications failed to improve her symptoms; however, the clinical manifestation was consistent with epilepsy with gait-induced seizures. Intracranial subdural EEG recording was performed and confirmed ictal activity originating from the right supplementary motor area. Resection of this area resulted in complete resolution of her symptoms.<br>
Discussion: This is the first reported case of successful resective surgery for epilepsy with gait-induced seizure. Brain networks involving cortical regions responsible for the initiation or execution of walking presumably played a key role in the generation of gait-induced seizures. Careful assessment using non-invasive neurophysiological studies facilitated accurate diagnosis, successful intracranial recordings, and effective resective surgery.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2328-95032025Dorsolateral Cervical Cord T2 Hyperintensity in KIF1C-Related Disease (Spastic Paraplegia 58): Two Long-Duration CasesENAkihikoMitsutakeDepartment of Neurology, Graduate School of Medicine, The University of TokyoMasaoOsakiDepartment of Neurology, Graduate School of Medicine, The University of TokyoTakashiMatsukawaDepartment of Neurology, Graduate School of Medicine, The University of TokyoMihoOsakoDepartment of Neurology, Tokyo Metropolitan Kita Medical and Rehabilitation Center for the DisabledChisenTakeuchiDepartment of Neurology, Tokyo Metropolitan Kita Medical and Rehabilitation Center for the DisabledHiroyukiIshiuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesJunMitsuiDepartment of Precision Medicine Neurology, Graduate School of Medicine, The University of TokyoRyoKurokawaDepartment of Radiology, Graduate School of Medicine, The University of TokyoHarushiMoriDepartment of Radiology, School of Medicine, Jichi Medical UniversityYujiTakahashiDepartment of Neurology, Graduate School of Medicine, The University of TokyoJunGotoDepartment of Neurology, Graduate School of Medicine, The University of TokyoShojiTsujiInstitute of Medical Genomics, International University of Health and WelfareTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of TokyoPathogenic variants in KIF1C cause Spastic Paraplegia 58 (SPG58), typically presenting with cerebellar ataxia and spastic paraparesis. We report two unrelated patients with spastic paraparesis, cerebellar ataxia, and tremor. Whole-exome sequence analysis identified novel homozygous variants in the motor domain of KIF1C (NM_006612.6): c.921G>A (p.Trp307Ter) and c.607C>T (p.Arg203Trp). In addition to the canonical brain MRI showing leukoencephalopathy with posterior dominance and hyperintensity along the corticospinal tracts, both patients showed symmetric T2 hyperintensity confined to the lateral and dorsal columns of the cervical cord. Given the long disease durations (22 and 51 years), these findings may represent late-emerging or previously overlooked spinal cord involvement and broaden the neuroradiological spectrum of SPG58.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama1085-94893042025A Case of Retinopathy–Sensory Neuropathy Syndrome With a Novel Compound Heterozygous FLVCR1 Variante70082ENYumikoNakanoDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYusukeFukuiDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKentaroDeguchiDepartment of Neurology, Okayama City General Medical CenterChikaMatsuokaDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomohitoKawanoDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiTairaDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAyakaMatsuoDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYosukeOsakadaDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTaijunYunokiDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesEmiNomuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMamiTakemotoDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyutaMoriharaDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesToruYamashitaDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHiroyukiIshiuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesBackground and Aims: Retinopathy–sensory neuropathy syndrome (RETSNS), also known as posterior column ataxia with retinitis pigmentosa (PCARP), is a rare neurodegenerative disorder that is caused by biallelic pathogenic variants in FLVCR1. Here, we report a case of a Japanese patient with RETSNS.<br>
Methods: Clinical, neuroradiological, and electrophysiological findings were documented. Whole-genome sequencing was performed. Subcloning was carried out to confirm compound heterozygosity. A functional assay was performed to assess the pathogenicity of the variants.<br>
Results: The patient showed retinitis pigmentosa and sensory ataxia. Over the course of the disease, autonomic dysfunction has become increasingly evident. Despite consanguinity in the family, whole-genome sequencing identified two heterozygous variants in FLVCR1 (c.369T>G, p.Phe123Leu and c.733A>G, p.Asn245Asp). Cloning of the PCR product followed by Sanger sequencing indicated compound heterozygosity of the variants. Immunocytochemistry of HEK293FT cells transfected with plasmids containing wild-type or variant FLVCR1 cDNA demonstrated altered subcellular localization of the variant FLVCR1 proteins, characterized by reduced membrane localization.<br>
Interpretation: We report a novel variant in FLVCR1 causing RETSNS. The functional assay supports the pathogenicity of the variants.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7962025Ileus Tube-Related Intussusception: A Case Report and Review of 80 Previously Reported Cases469474ENTeruyukiTsujiiDepartment of Surgery, Matsuda HospitalTatsuoMatsudaDepartment of Surgery, Matsuda HospitalYujiKimuraDepartment of Surgery, Matsuda HospitalRyoichiKatsubeDepartment of Surgery, Matsuda HospitalHironoriIwadouDepartment of Surgery, Matsuda HospitalSadamiFunabikiDepartment of Surgery, Matsuda HospitalYasuakiKamikawaDepartment of Surgery, Matsuda HospitalTadakazuMatsudaDepartment of Surgery, Matsuda HospitalCase Report10.18926/AMO/69851We report a rare case of ileus tube-related intussusception in an adult. A 56-year-old man with adhesive bowel obstruction was treated with a nasointestinal ileus tube. Although his condition initially improved, persistent abdominal pain led to the diagnosis of intussusception via CT imaging. Manual repositioning of the tube resolved the intussusception without the need for bowel resection. A review of 80 previously reported cases of ileus tube-associated intussusception (total 81 cases, 95 lesions) highlighted the timing of onset, treatment strategies, and precautions. Early detection and diagnosis are crucial to prevent severe complications and preserve bowel function.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7962025MRI Images of a Case of Adenocarcinoma, Human Papillomavirus-Independent, Mesonephric Type, of the Uterine Cervix463468ENYudaiAsanoDepartment of Radiology, Okayama University HospitalChikaNishiharaDepartment of Radiology, Okayama University HospitalTakahiroKitayamaDepartment of Radiology, Okayama University HospitalNanakoOkawaDepartment of Radiology, Okayama University HospitalSatokoMakimotoDepartment of Radiology, Okayama University HospitalFumiyoHigakiDepartment of Radiology, Okayama University HospitalKatsuhideKojimaDepartment of Radiology, Okayama University HospitalHanakoSugiharaDepartment of Obstetrics and Gynecology, Okayama University HospitalNaoyukiIdaDepartment of Obstetrics and Gynecology, Okayama University HospitalHiroyukiYanaiDepartment of Pathology, Okayama University HospitalTakaoHirakiDepartment of Radiology, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityCase Report10.18926/AMO/69850We present a case of a woman in her 70s who was diagnosed with mesonephric adenocarcinoma of the uterine cervix, following biopsy and surgery. Preoperative MRI revealed a 7-cm, well-defined circumferential cervical mass with left lateral wall predominance, bulging into the uterine cavity and vagina. The lesion showed intermediate signal intensity on T2-weighted images, diffusion restriction, and early contrast enhancement weaker than that of the myometrium, followed by washout on contrast-enhanced imaging. The circumferential growth pattern with the lateral wall predominance and its imaging characteristics may suggest this rare entity be routinely included in the differential diagnosis of cervical cancers.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7962025Exacerbation of Proteinuria in a Patient with Behçet’s Disease and IgA Nephropathy Following Colchicine Discontinuation457461ENTomohikoAsakawaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesHaruhito A.UchidaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesYuKatayamaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesYoshimasaSakurabuDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKatsuyoshiKatayamaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesYasuhiroOnishiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesNatsumiMatsuoka-UchiyamaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesHidemiTakeuchiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKeikoTanakaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKenjiTsujiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesRyokoUmebayashiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesRikaTakemotoDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/69849This case involves a 23-year-old male who was diagnosed with Behçet’s disease 5 years ago and managed with colchicine. Two months ago, he underwent renal biopsy due to abnormal urinalysis and kidney dysfunction, leading to a diagnosis of IgA nephropathy. He subsequently underwent tonsillectomy followed by glucocorticoid pulse therapy. However, after the tonsillectomy, discontinuing colchicine led to increased proteinuria, despite the glucocorticoid pulse therapy. Upon reintroducing colchicine, urinary protein excretion decreased, achieving incomplete remission. These findings suggest that colchicine may be effective in decreasing proteinuria in patients with Behçet’s disease complicated by IgA nephropathy.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7962025Recurrence of FVIII Inhibitor during Surgery in a Patient with Severe Hemophilia A Receiving Emicizumab Prophylaxis451455ENMoeHagiharaDepartment of Hematology and Oncology, Okayama University HospitalKeisukeSeikeDepartment of Hematology and Oncology, Okayama University HospitalKentaHayashinoDepartment of Hematology and Oncology, Okayama University HospitalTakaoYasuharaDepartment of Neurological Surgery, Okayama University HospitalKyoheiKinDepartment of Neurological Surgery, Okayama University HospitalYuichiHirataDepartment of Neurological Surgery, Okayama University HospitalHirokiKobayashiDepartment of Hematology and Oncology, Okayama University HospitalWataruKitamuraDepartment of Hematology and Oncology, Okayama University HospitalHideakiFujiwaraDepartment of Hematology and Oncology, Okayama University HospitalNoboruAsadaDepartment of Hematology and Oncology, Okayama University HospitalNobuharuFujiiDivision of Transfusion and Cell Therapy, Okayama University HospitalYoshinobuMaedaDepartment of Hematology and Oncology, Okayama University HospitalCase Report10.18926/AMO/69848Emicizumab, a bispecific monoclonal antibody, benefits patients with severe hemophilia A. It alters laboratory assessments of coagulation activity, requiring anti-idiotype monoclonal antibodies for accurate monitoring. A 64-year-old man, receiving emicizumab regularly, was admitted for laminoplasty. We planned to use FVIII replacement during the perioperative period after confirming the disappearance of inhibitors, monitoring coagulation activity with anti-idiotype monoclonal antibodies. Activated partial thromboplastin time was prolonged on postoperative day 2, prompting an immediate switch to eptacog alfa. The patient recovered without bleeding. This case underscores the necessity of anti-idiotype monoclonal antibodies for accurate monitoring.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2666-084930322025Guide Tip Damage Due to Rotablation105347ENSatoshiTayaDepartment of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical ScienceMasatokiYoshidaDepartment of Cardiovascular Medicine, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKentaroEjiriDepartment of Cardiovascular Medicine, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesHironobuTodaDepartment of Cardiovascular Medicine, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKazufumiNakamuraDepartment of Cardiovascular Medicine, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesHiroshiMoritaDepartment of Cardiovascular Medicine, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesShinsukeYuasaDepartment of Cardiovascular Medicine, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesBackground: The rotational atherectomy system can effectively debulk calcified coronary lesions. However, rare complications specific to that system have been reported.<br>
Case Summary: A 77-year-old man with a heavily calcified lesion in the right coronary artery (RCA) ostium underwent percutaneous coronary intervention in an 8-F system. During the procedure, rotablation with a 2.25-mm burr was required. After the percutaneous coronary intervention, partial loss of the tip of the guide was observed. He had no clinical sequelae despite the presumed retained catheter material in his body.<br>
Discussion: Although insufficient guide coaxiality has been suggested as the primary cause of guide tip fracture during RCA ostial ablation, other factors may have contributed: the application of force to the tip and a small difference in size between the guide and the burr.<br>
Take-Home Message: When ablating RCA ostial lesions, positioning the burr platform outside the guide may help prevent similar complications in future cases.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-090413122025Focal Nodular Hyperplasia of the Liver Mimicking Gastric Submucosal Tumore71666ENYoheiMasudaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshiakiSoejimaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFumioOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFocal nodular hyperplasia (FNH) is a common benign hepatic tumor, typically featuring a central scar with a spoke-wheel pattern on abdominal ultrasound. This case highlights a rare presentation of FNH causing extragastric compression that mimicked a gastric submucosal tumor on esophagogastroduodenoscopy.No potential conflict of interest relevant to this article was reported.MDPI AGActa Medica Okayama2077-038314142025Symptomatic Trends and Time to Recovery for Long COVID Patients Infected During the Omicron Phase4918ENHiroshiAkiyamaDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYasueSakuradaDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityHiroyukiHondaDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYuiMatsudaDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYukiOtsukaDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKazukiTokumasuDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYasuhiroNakanoDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityRyosukeTakaseDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityDaisukeOmuraDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKeigoUedaDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityFumioOtsukaDepartment of General Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityBackground: Since the pathophysiology of long COVID is not yet fully understood, there are no specific methods for its treatment; however, its individual symptoms can currently be treated. Long COVID is characterized by symptoms that persist at least 2 to 3 months after contracting COVID-19, although it is difficult to predict how long such symptoms may persist. Methods: In the present study, 774 patients who first visited our outpatient clinic during the Omicron period from February 2022 to October 2024 were divided into two groups: the early recovery (ER) group (370 cases; 47.8%), who recovered in less than 180 days (median 33 days), and the persistent-symptom (PS) group (404 cases; 52.2%), who had symptoms that persisted for more than 180 days (median 437 days). The differences in clinical characteristics between these two groups were evaluated. Results: Although the median age of the two groups did not significantly differ (40 and 42 in ER and PS groups, respectively), the ratio of female patients was significantly higher in the PS group than the ER group (59.4% vs. 47.3%). There were no significant differences between the two groups in terms of the period after infection, habits, BMI, severity of COVID-19, and vaccination history. Notably, at the first visit, female patients in the PS group had a significantly higher rate of complaints of fatigue, insomnia, memory disturbance, and paresthesia, while male patients in the PS group showed significantly higher rates of fatigue and headache complaints. Patients with more than three symptoms at the first visit were predominant in the PS groups in both genders. Notably, one to two symptoms were predominant in the male ER group, while two to three symptoms were mostly reported in the female PS group. Moreover, the patients in the PS group had significantly higher scores for physical and mental fatigue and for depressive symptoms. Conclusions: Collectively, these results suggest that long-lasting long COVID is related to the number of symptoms and presents gender-dependent differences.No potential conflict of interest relevant to this article was reported.Oxford University Press (OUP)Acta Medica Okayama2042-88122025122025Endoscopic surgery for distal femoral physeal bar resection with computed tomography-assisted navigation: a case reportrjaf972ENYasutakaMasadaDepartment of Orthopaedic Surgery, Science of Functional Recovery and Reconstruction, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomonoriTetsunagaDepartment of Musculoskeletal Health Promotion, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKazukiYamadaDepartment of Medical Materials for Musculoskeletal Reconstruction, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTomohiroInoueDepartment of Orthopaedic Surgery, Science of Functional Recovery and Reconstruction, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyuichiroOkudaDepartment of Orthopaedic Surgery, Science of Functional Recovery and Reconstruction, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTetsuyaYamamotoDepartment of Orthopaedic Surgery, Science of Functional Recovery and Reconstruction, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinMatsumotoDepartment of Orthopaedic Surgery, Science of Functional Recovery and Reconstruction, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomokoTetsunagaDepartment of Sports Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYusukeYokoyamaDepartment of Advanced Rehabilitation Medicine for the Musculoskeletal System, Okayama UniversityYukiOkazakiCenter for Education in Medicine and Health Sciences, Okayama UniversityToshifumiOzakiDepartment of Orthopaedic Surgery, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityThe formation of physeal bars, or bony bridges, following growth plate injuries can cause complications such as angular deformities or discrepancies in leg length. The management strategies for these depend on factors such as the bar’s location, extent, and residual growth potential. Herein, we describe the case of a 14-year-old male with a valgus knee deformity caused by a distal femoral physeal bar. The patient underwent endoscopic resection of the bar, assisted by computed tomography-based navigation and intraoperative O-arm imaging. This minimally invasive technique facilitated safe and accurate removal of the lesion with less risk of complications such as cortical perforation or injury to adjacent neurovascular structures compared to traditional approaches. The patient experienced favorable postoperative outcomes, including restored knee range of motion and full symptom resolution. This approach demonstrates the clinical value of integrating endoscopy with advanced navigation systems during the surgical treatment of physeal bars.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2210-26121342025Robotic posterior radical antegrade modular pancreatosplenectomy for left-sided pancreatic cancer using the ligament of Treitz first approach: A case report and technical note111782ENKoseiTakagiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomokazuFujiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuyaYasuiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuyukiMatsumotoDepartment of Gastroenterology, Okayama University HospitalToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesIntroduction: Radical antegrade modular pancreatosplenectomy (RAMPS) is the standardized open surgical technique for treating left-sided pancreatic cancer. However, studies reporting the surgical approaches for robotic RAMPS are limited. Here, we present a robotic posterior RAMPS using the ligament of Treitz first approach.<br>
Presentation of case: A 46-year-old male patient with initially unresectable pancreatic body cancer underwent robotic posterior RAMPS as a conversion surgery after 1-year of chemotherapy with modified FOLFIRINOX.<br>
Discussion: Following evaluation of resectability, the ligament of Treitz first approach was applied. The transverse colon was lifted cranially, and the left renal vein was exposed after dissection around the ligament of Treitz. The left adrenal vein was divided, and the left adrenal gland was resected with special caution to avoid injury to the left renal artery. Retroperitoneal dissection was performed with lymphadenectomy around the superior mesenteric and celiac arteries using the ligament of Treitz first approach. After repositioning the transverse colon, the gastrocolic and gastrosplenic ligaments were dissected. Following the division of the pancreas and splenic vessels, the retroperitoneal dissection line was connected with those of the ligament of Treitz first approach. The operative time was 303 min, and the estimated blood loss was 150 mL.<br>
Conclusion: The ligament of Treitz first approach may be an option for robotic RAMPS for left-sided pancreatic cancer. Surgeons should select the best approach for performing robotic RAMPS.No potential conflict of interest relevant to this article was reported.Japanese Society of Internal MedicineActa Medica Okayama0918-291864232025Remarkable Efficacy of Capmatinib in a Patient with Cancer of Unknown Primary with MET Amplification34603464ENTakaakiTanakaDepartment of Respiratory Medicine, Okayama University HospitalGoMakimotoDepartment of Respiratory Medicine, Okayama University HospitalRyoheiSumiiDepartment of General Internal Medicine, NHO Fukuyama Medical CenterRikaOmoteDepartment of Pathology, NHO Fukuyama Medical CenterYayoiAndoClinical Research Support Office, National Cancer Center HospitalKiichiroNinomiyaCenter for Comprehensive Genomic Medicine, Okayama University HospitalEikiIchiharaCenter for Clinical Oncology, Okayama University HospitalKadoakiOhashiDepartment of Respiratory Medicine, Okayama University HospitalYoshinobuMaedaDepartment of Hematology, Oncology, and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMasahiroTabataCenter for Clinical Oncology, Okayama University HospitalThis case report describes a 70-year-old female with cancer of unknown primary origin (CUP) who exhibited multiple distant lymph node metastases. Despite conventional chemotherapy (carboplatin and paclitaxel) and immunotherapy (nivolumab), disease progression was noted. Genomic profiling revealed MET amplification, leading to the administration of capmatinib, a selective MET tyrosine kinase inhibitor. The patient experienced substantial tumor reduction with dose adjustments due to adverse effects, indicating the potential efficacy of capmatinib in treating CUP with MET amplification.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1865-72572025Avoiding splenectomy in splenic sclerosing angiomatoid nodular transformation through endoscopic ultrasound-guided tissue acquisition: a 36-month follow-up case reportENTakakiOkuyamaDepartment of Internal Medicine, Tsuyama Chuo HospitalKazuyukiMatsumotoDepartment of Gastroenterology and Hepatology, Okayama University HospitalKosakuMorimotoDepartment of Internal Medicine, Tsuyama Chuo HospitalShogoKimuraDepartment of Internal Medicine, Tsuyama Chuo HospitalTakayoshiMiyakeDepartment of Pathology, Tsuyama Chuo HospitalTakuyaSatomiDepartment of Internal Medicine, Tsuyama Chuo HospitalKensukeTakeiDepartment of Internal Medicine, Tsuyama Chuo HospitalShogoInoueDepartment of Internal Medicine, Tsuyama Chuo HospitalRyutaTakenakaDepartment of Internal Medicine, Tsuyama Chuo HospitalA 48-mm splenic mass was incidentally discovered in a 78-year-old man upon computed tomography. Follow-up imaging at 12 months revealed enlargement to 60 mm, prompting endoscopic ultrasound-guided tissue acquisition with a 22-gauge needle. Histopathological analysis confirmed that it was a sclerosing angiomatoid nodular transformation. The patient was asymptomatic and had no hematologic abnormalities; therefore, splenectomy was not performed. After biopsy, the lesion regressed from 60 mm to 46 mm, possibly owing to hematoma formation or vascular disruption, and remained stable during 36 months of follow-up. Although splenectomy has been performed in most reported cases of sclerosing angiomatoid nodular transformation because of diagnostic uncertainty, a few recent reports have demonstrated that sclerosing angiomatoid nodular transformation can be diagnosed by endoscopic ultrasound-guided tissue acquisition, thereby avoiding splenectomy. This case highlights the diagnostic utility of endoscopic ultrasound-guided tissue acquisition and supports spleen-preserving management for biopsy-proven sclerosing angiomatoid nodular transformation.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1433-73982025A rare case of supratentorial ependymosarcoma harboring ZFTA::RELA fusionENYujiMatsumotoDepartment of Neurological Surgery, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYasukiSurugaDepartment of Neurological Surgery, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKaishiSatomiDepartment of Pathology, Faculty of Medicine, Kyorin UniversityYoheiInoueDepartment of Neurological Surgery, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYasuhikoHattoriDepartment of Neurological Surgery, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineJojiIshidaDepartment of Neurological Surgery, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKazuhikoKurozumiDepartment of Neurosurgery, Hamamatsu University HospitalSumihitoNobusawaDepartment of Human Pathology, Gunma University School of MedicineJunkoHiratoDepartment of Pathology, Public Tomioka General HospitalTakehiroTanakaDepartment of Pathology, Okayama University HospitalHiroyukiYanaiDepartment of Pathology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKanaWashioDepartment of Pediatrics, Okayama University HospitalKoichiIchimuraDepartment of Pathology, Faculty of Medicine, Kyorin UniversityTomotsuguIchikawaDepartment of Neurosurgery, Kagawa Prefectural Central HospitalYoshihiroOtaniDepartment of Neurological Surgery, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShotaTanakaDepartment of Neurological Surgery, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineEpendymosarcoma is an exceedingly rare variant of ependymoma characterized by a mixture of ependymomatous and sarcomatous components. We report a case of supratentorial ependymosarcoma harboring a ZFTA::RELA fusion in a 10-year-old girl. Histologically, the tumor comprised an ependymomatous component resembling clear cell ependymoma and a sarcomatous component. ZFTA::RELA fusion was confirmed in both components. Genome-wide methylation profiling classified both components as supratentorial ependymoma, ZFTA fusion–positive by the German Cancer Research Center (DKFZ) CNS tumor classifier v12b8. However, their copy number alteration profiles were distinct. The ependymomatous component exhibited a gain of chromosome 1q and a loss of chromosomes 1p, 9, and 19q, while the sarcomatous component showed a loss of chromosome 14. These findings suggest that both components may have differentiated from a common precursor despite their distinct morphologies. The patient underwent gross total resection followed by adjuvant chemoradiotherapy and remains recurrence-free eight years post-treatment. Further investigation of additional cases is warranted to better understand the pathogenesis of this rare tumor.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841782025Conversion to Hip Arthroplasty After Internal Fixation Failure in an Intertrochanteric Femoral Fracture: A Case Reporte90112ENShiroFukuokaDepartment of Orthopedics, Kagawa Prefectural Central HospitalTomooInoueDepartment of Orthopedics, Kagawa Prefectural Central HospitalMotokiTakahashiDepartment of Orthopedics, Kagawa Prefectural Central HospitalKeisukeKawasakiDepartment of Orthopedic Surgery, Kagawa Prefectural Central HospitalToshifumiOzakiDepartment of Orthopedic Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical ScienceIntertrochanteric femoral fractures are mainly managed by internal fixation. However, failures such as over-telescoping, cut-out, nonunion, or implant failure can occur, especially in osteoporotic elderly patients. We report the case of a patient in whom we performed artificial hip replacement surgery after fixation failure following internal fixation of an intertrochanteric femoral fracture. We report the case of an 85-year-old woman who sustained a left intertrochanteric femoral fracture treated with a dynamic hip screw (DHS). One week postoperatively, radiographs revealed over-telescoping of the lag screw. The patient did not complain of pain, but she underwent conversion to cemented bipolar hemiarthroplasty under general anesthesia. One possible cause of over-telescoping of the lag screw after surgery was that the longitudinal fracture line in the calcar of the proximal bone fragment, as seen in the initial CT image, may have extended horizontally at the neck level. During surgery, a fracture at the same site caused the anterior medial fragment to fail, resulting in a coronal shear fracture and fixation failure. When a longitudinal fracture line is observed in the calcar of the proximal fragment, it is necessary to keep in mind that it may extend horizontally at the neck level.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841732025A Case of Charcot Spine Arthropathy at the Lumbosacral Level in a Patient With Ankylosis of the Spinee80656ENYoshiakiOdaDepartment of Orthopedic Surgery, Okayama University HospitalKojiUotaniDepartment of Musculoskeletal Traumatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTomokoTetsunagaDepartment of Orthopedic Surgery, Okayama University HospitalKensukeShinoharaDepartment of Orthopedic Surgery, Okayama University HospitalToshifumiOzakiDepartment of Orthopedic Surgery, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityCharcot spinal arthropathy, a rare refractory progressive disease, is characterized by symptoms such as pain, deformity, and neurological impairment, which can significantly reduce functional ability, quality of life, and life expectancy. We report a case of Charcot spine at the L5/S1 level with long segment ankylosis to the L5 vertebra. We first performed thorough debridement via a posterior approach. We used antibiotic-containing cement as a spacer to fill the dead space, facilitating the second surgery approach. In the second surgery, transdiscal screws, which have a low profile and strong force, were used as anchors, and bulk bone harvested from both iliac bones was grafted to the intervertebral space. The lumbosacral alignment was kyphotic, and the patient could sit and move independently. Disimpaction was impossible, and a stoma had to be created.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2045-23221512025Elucidation of puberulic acid–induced nephrotoxicity using stem cell-based kidney organoids42195ENHiroyukiNakanohDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesKenjiTsujiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesNaruhikoUchidaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesKazuhikoFukushimaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesSoichiroHaraguchiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesShinjiKitamuraDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesRecent cases of acute kidney injury (AKI) in Japan have been linked to Beni-koji CholesteHelp supplements, with puberulic acid identified as a potential nephrotoxic contaminant. To address the need for a reliable in vitro nephrotoxicity testing platform, we developed a screening model using kidney organoids derived from adult rat kidney stem (KS) cells. The organoids were exposed to known nephrotoxicants, including cisplatin and gentamicin, to validate the system. Puberulic acid toxicity was evaluated in both KS cell-derived organoids and wild-type mice. The organoids recapitulated tubular injury induced by known nephrotoxins and showed significant Kim-1 mRNA upregulation. Puberulic acid-treated organoids and mice exhibited morphological features of acute tubular necrosis (ATN), mitochondrial damage, and reduced cytochrome c oxidase subunit IV (COX-IV) expression. Markers of oxidative stress and apoptosis, such as 8-hydroxy-2’-deoxyguanosine (8-OHdG) and cleaved caspase-3, were also elevated. These findings suggest that puberulic acid induces mitochondrial dysfunction and oxidative stress, leading to tubular cell death. Puberulic acid-induced nephrotoxicity was demonstrated using our kidney organoid model. KS cell-derived kidney organoids may provide a simple, reproducible, and rapid platform for nephrotoxicity assessment, which may complement conventional animal experiments.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama1328-80676712025Pediatric stroke risk and neurotrauma from roller coasters in amusement parkse70221ENTomokiMorikawaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTakafumiObaraDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTsuyoshiNojimaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityKoheiTokiokaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityAtsunoriNakaoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityKoheiTsukaharaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityAlthough rare, neurotrauma has been documented as a potential risk of high-speed, high-acceleration amusement park rides such as roller coasters. These attractions generate rapid acceleration, deceleration, sharp turns, and significant gravitational forces, which may stress the central nervous system and cerebrovascular structures. This review analyzed pediatric stroke cases (children 15 years old or younger) linked to roller-coaster rides reported in PubMed and summarized the key mechanisms and clinical features associated with such neurotrauma. Documented complications include internal and vertebral carotid artery dissections, with or without stroke, subdural hemorrhage, intraparenchymal hemorrhage, and post-traumatic migraines. The aim of this review is to alert healthcare providers to the possibility of stroke induced by roller-coaster rides, emphasizing the importance of timely diagnosis and management to prevent adverse outcomes. Key considerations include the recognition of risk factors, public education on potential risks, and strategies for preventing complications in at-risk populations. Although intracranial hemorrhage from roller-coaster rides is rare, individuals with predisposing conditions, such as prior head trauma or vascular abnormalities, should be evaluated carefully when presenting with neurological symptoms after such activities.No potential conflict of interest relevant to this article was reported.Japan Medical AssociationActa Medica Okayama2433-3298822025Successful Transplantation of Multiple Organs from Donor after Helium Asphyxiation: First Case Report in Japan650653ENShuntaJinnoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTakashiHongoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTakafumiObaraDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTsuyoshiNojimaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityKoheiTsukaharaDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTetsuyaYumotoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityHiromichiNaitoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityAtsunoriNakaoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityHelium inhalation has increased, but most cases are either minor injuries or deaths; there have not yet been any reported cases of brain death leading to organ donation. We report a patient who attempted helium inhalation and was declared brain dead and became an organ donor without complications. To the best of our knowledge, this is the first reported case of deceased organ donation following helium asphyxiation in Japan. The patient in cardiac arrest was found with a helium-filled vinyl bag sealed around the neck. During emergency medical transport to the hospital, a spontaneous return of circulation was obtained after 31 minutes of cardiopulmonary resuscitation. Upon hospital arrival, the physical examination revealed dilated pupils with no response to light. Electrocardiography showed widespread ST-segment depression and ST-segment elevation in augmented Vector Right, as well as elevated cardiac enzymes and decreased myocardial contractility. Head computed tomography revealed diffuse cerebral edema and loss of the gray-white matter boundary without signs of air embolism in the cerebral and coronary arteries. Despite comprehensive post-cardiac arrest care with recovery of organ function, brain death was confirmed on day 4 after hospitalization. The family consented to organ donation on the 11th day of hospitalization. The heart, lungs, liver, and two kidneys were successfully transplanted and all organs functioned. All organ grafts were functioning well at the 3-month follow-up. Our case demonstrates that brain death caused by helium inhalation is not a contraindication to organ donation.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1865-13801812025Tattoo-associated toxic shock syndrome: a case report185ENTakuyaKuboDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTetsuyaYumotoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalKojiIioMicrobiology Division, Clinical Laboratory, Okayama University HospitalHiromichiNaitoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityAtsunoriNakaoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityBackground: Toxic shock syndrome (TSS) is a rare but life-threatening complication occasionally reported after tattooing.<br>
Case presentation: : A 29-year-old Japanese man was admitted to Okayama University Hospital, Okayama, Japan, in early spring 2025, one week after receiving a tattoo on his right shoulder and upper arm in Osaka. He presented with fever, gastrointestinal symptoms, hypotension, and multi-organ failure. Despite a failure to isolate a causative pathogen, he met clinical criteria for TSS. Supportive care and broad-spectrum antibiotics led to full recovery.<br>
Conclusions: TSS can occur after tattooing, even in individuals without apparent immunodeficiency. Pathogenic organisms may be unidentifiable; however, clinical diagnosis should not be delayed, and early therapeutic interventions are essential to improve outcomes.No potential conflict of interest relevant to this article was reported.Ivyspring International PublisherActa Medica Okayama1837-966416142025CXCR4 Inhibition Induces Tumor Necrosis by Selectively Targeting the Proliferating Blood Vessels in Oral Squamous Cell Carcinoma40554070ENYaminSoeDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Science, Okayama UniversityHotakaKawaiDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Science, Okayama UniversityHtoo ShweEainDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Science, Okayama UniversitySaoriYoshidaPreliminary Examination Room, Okayama University HospitalMay WathoneOoDepartment of Pathophysiology and Drug Discovery, Graduate School of Medicine, Dentistry and Pharmaceutical Science, Okayama UniversityZin ZinMinDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Science, Okayama UniversityKiyofumiTakabatakeDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Science, Okayama UniversityKeisukeNakanoDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Science, Okayama UniversityHitoshiNagatsukaDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Science, Okayama UniversityThe C-X-C chemokine receptor type 4 (CXCR4) is a G protein-coupled transmembrane receptor that contributes to tumor growth and angiogenesis. While prior studies have primarily focused on CXCR4 expression in cancer cells and its role in metastasis, a few have examined its involvement in tumor-associated vasculature. In this study, we reported for the first time that CXCR4 expression within the tumor vasculature is significantly associated with higher pathological grades of human oral squamous cell carcinoma (OSCC) (p<0.03). A previous study reported that inhibiting CXCR4 with AMD3100 induces tumor cell death and enhances the efficacy of the chemotherapeutic agent cisplatin. These findings suggest that CXCR4 is an important target for cancer treatment. However, the tumor vascular system is known to be heterogeneous within the tumor microenvironment (TME), which may influence the treatment outcomes. Therefore, this study aimed to explore the effect of CXCR4 antagonism on various blood vessels present within the oral squamous cell carcinoma (OSCC) tumor stroma. Although the efficiency of AMD3100 was not significant in MOC cancer cells, necrosis was induced in the TME when applied to a poorly differentiated OSCC model, highlighting the role of the TME. Notably, CXCR4 is found to be highly overlapped with CD105+ angiogenic tumor vessels among various vascular markers. Treatment with AMD3100 leads to a marked reduction in the CD105+ vessels and impairs the maturation of tumor micro-vessels, explaining the cause of observed necrosis. Thus, CXCR4 serves as a promising biomarker in OSCC, and its inhibition with AMD3100 offers the therapeutic potential, particularly in cases with advanced pathological grades.No potential conflict of interest relevant to this article was reported.BioscientificaActa Medica Okayama2052-0573202512025Adult hypophosphatasia presenting with recurrent acute joint paine240121ENHayaoYoshidaDepartment of Diabetes and Endocrinology, Shiga General HospitalTakaakiMurakamiDepartment of Diabetes and Endocrinology, Shiga General HospitalAtsubumiOgawaDepartment of Rheumatology and Clinical Immunology, Kyoto University Graduate School of MedicineTakashiSunouchiDivision of Nephrology and Endocrinology, The University of Tokyo HospitalNaokoHidakaDivision of Nephrology and Endocrinology, The University of Tokyo HospitalNobuakiItoOsteoporosis Center, The University of Tokyo HospitalHiromiMurakamiDepartment of Genomic Medicine, Kyoto University Graduate School of MedicineHidenoriKawasakiDepartment of Genomic Medicine, Kyoto University Graduate School of MedicineTomoyukiAkiyamaDepartment of Child Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKatsumiNakajimaDepartment of Diabetes and Endocrinology, Shiga General HospitalDaisukeYabeDepartment of Diabetes, Endocrinology and Nutrition, Kyoto University Graduate School of MedicineTaizoYamamotoDepartment of Diabetes and Endocrinology, Shiga General HospitalHypophosphatasia (HPP) is a genetic disorder due to pathological variants in ALPL, the gene encoding tissue-nonspecific alkaline phosphatase (ALP). HPP is typically associated with bone-related symptoms, such as bone deformity, fractures and bone pain in children, but can appear in adults with symptoms resembling arthritis. A 22-year-old male experienced repeated and severe sudden attacks of joint pain in the elbows and knees. Magnetic resonance imaging and joint ultrasonography showed joint effusions indicating chronic inflammation. Blood biochemical tests revealed a remarkably low serum ALP level, and repeated examination confirmed a sustained low ALP level; urine phosphoethanolamine, plasma inorganic pyrophosphate and plasma pyridoxal-5′-phosphate levels were elevated, raising concern for HPP. While the patient had no history of premature loss of primary teeth, fragility fractures, muscle weakness or abnormalities in growth, genetic testing revealed a likely pathogenic and a pathogenic heterozygous variant in the ALPL gene, i.e., c.979T>C (p.Phe327Leu) and c.1559del (p.Leu520Argfs), confirming HPP. Additional genetic testing of his parents showed a heterozygous c.1559del variant in his father and a heterozygous c.979T>C variant in his mother. A diagnosis of adult HPP due to compound heterozygous mutations was therefore confirmed. Enzyme replacement therapy with asfotase alfa was then introduced; no attacks of arthralgia occurred in the 1-year period since then. This case highlights the possibility of HPP in adults who present clinically with repeated joint symptoms and low serum ALP levels but without bone-related symptoms.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2688-6146652025Late‐Onset Invasive Aspergillosis With Pituitary Involvement and Dysfunction Following CD19 Chimeric Antigen Receptor T‐Cell Therapye70138ENDaisukeIkedaDepartment of Hematology, Oncology and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomohiroNawadaThe Center for Graduate Medical Education, Okayama University HospitalTakumiKondoDepartment of Hematology and Oncology, Okayama University HospitalTakayukiShinoharaDepartment of Fungal Infection, National Institute of Infectious DiseasesTomohiroNaganoDepartment of Hematology and Oncology, Okayama University HospitalSayaKubotaDepartment of Hematology, Oncology and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyuichiroHiyamaDepartment of Hematology and Oncology, Okayama University HospitalMasayaUenoDepartment of Hematology and Oncology, Okayama University HospitalHirokiKobayashiDepartment of Hematology and Oncology, Okayama University HospitalKeisukeSeikeDepartment of Hematology and Oncology, Okayama University HospitalHideakiFujiwaraDepartment of Hematology and Oncology, Okayama University HospitalNoboruAsadaDepartment of Hematology and Oncology, Okayama University HospitalDaisukeEnnishiDepartment of Hematology and Oncology, Okayama University HospitalKeikoFujiiDepartment of Hematology and Oncology, Okayama University HospitalNobuharuFujiiDepartment of Hematology and Oncology, Okayama University HospitalMasanoriMakitaDepartment of Hematology, Chugoku Central HospitalYoshinobuMaedaDepartment of Hematology and Oncology, Okayama University HospitalIntroduction: Invasive fungal infection (IFI) after chimeric antigen receptor (CAR) T-cell therapy is less common than bacterial and viral infections, but can be fatal once it develops. As most cases occur within 30 days after CAR T-cell infusion, late-onset IFI―particularly mould infection―appears to be under-recognised.<br>
Discussion: We report an illustrative case of pituitary aspergillosis developing as late as one year after CD19 CAR T-cell therapy, highlighting a persistent risk in certain patients with delayed immune reconstitution.<br>
Conclusion: This case underscores the need for continued vigilance and individualised antifungal strategies to prevent IFI beyond the early post-infusion period.<br>
Trial Registration: The authors have confirmed clinical trial registration is not needed for this submission.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1436-32912862025Gallbladder edema as a clue to zolbetuximab-associated protein-losing enteropathy in gastric cancer: a case report12971301ENYoshihikoKakiuchiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShinjiKurodaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShunyaHanzawaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineNobuhikoKanayaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineHajimeKashimaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineSatoruKikuchiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKunitoshiShigeyasuDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYoshiyasuKonoDepartment of Gastroenterology and Hepatology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShunsukeKagawaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineWe report a rare case of protein-losing enteropathy (PLE) during zolbetuximab treatment in a 73-year-old woman with Stage IVB gastric cancer. After chemo-immunotherapy and curative surgery, 3rd-line treatment with capecitabine, oxaliplatin, and zolbetuximab was initiated due to recurrence. The patient developed persistent right upper abdominal pain; imaging revealed gallbladder wall edema, followed by mild gastric wall edema, despite unremarkable laboratory findings. Protein-losing scintigraphy demonstrated abnormal gastric protein leakage, leading to a diagnosis of PLE. While gastrointestinal toxicity is known with zolbetuximab, this is, to our knowledge, the first clinically diagnosed case of PLE in which gallbladder edema served as a diagnostic clue. As treatment strategies for advanced gastric cancer grow increasingly complex, achieving maximum therapeutic benefit requires not only optimal drug selection but also timely recognition and management of adverse events. With the broader use of zolbetuximab, clinicians should be mindful of this rare but potentially significant complication.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1279-85174712025A case of a large venous ring around the mandibular condyle95ENKeitaroNishiDepartment of Maxillofacial Diagnostic and Surgical Science, Field of Oral and Maxillofacial Rehabilitation, Graduate School of Medical and Dental Sciences, Kagoshima UniversityTatsuoOkuiDepartment of Maxillofacial Diagnostic and Surgical Science, Field of Oral and Maxillofacial Rehabilitation, Graduate School of Medical and Dental Sciences, Kagoshima UniversityYoheiTakeshitaDepartment of Oral and Maxillofacial Radiology, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityJingoKusukawaDental and Oral Medical Center, Kurume University School of MedicineR. ShaneTubbsDepartment of Neurosurgery, Clinical Neuroscience Research Center, Tulane University School of MedicineJoeIwanagaDental and Oral Medical Center, Kurume University School of MedicineAnatomical details regarding venous drainage of the head and neck are an important matter for surgeons to avoid unnecessary complications such as hemorrhage. This report describes a case of the large venous ring around the mandibular condyle found in the cadaver. The left maxillofacial region of a latex-injected embalmed male cadaver (82 years of age at death) was dissected. The large two maxillary veins ran lateral to the capsule and superior to the mandibular notch and coursed posteroinferiorly to merge, and one trunk was formed at the posterior border of the ramus. It then received the superficial temporal vein superiorly to form the retromandibular vein (RMV). In addition, three maxillary veins were drained from the pterygoid venous plexus (PVP), medial to the ramus, one maxillary vein drained from the PVP into the RMV trunk, while two maxillary veins drained from the PVP into the anterior division of the RMV. All five large veins lateral and medial to the condyle drained from the PVP into the RMV. The knowledge of such an anatomical variation might prevent intraoperative bleeding in the temporomandibular joint region.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2363-90241112025Venous air embolism induced by burr hole drilling before dural incision in craniotomy: two case reports59ENYoheiMotoiDepartment of Anesthesiology and Resuscitology, Okayama University HospitalShujiOkaharaDepartment of Anesthesiology and Resuscitology, Okayama University HospitalMakikoTaniDepartment of Anesthesiology and Resuscitology, Okayama University HospitalNobushigeTsuboiDepartment of Neurosurgery, Okayama Red Cross HospitalHiroshiMorimatsuDepartment of Anesthesiology and Resuscitology, Okayama University HospitalBackground: Venous air embolism (VAE) is a rare but potentially fatal complication in neurosurgery typically caused by injury to dura mater, especially venous sinuses, during craniotomy. We report two cases of VAE that occurred before dural incision.<br>
Case presentation: Both patients underwent craniotomy under general anesthesia in a head-up position. Hemodynamic and respiratory deterioration occurred during or immediately after burr hole drilling with abnormal vital signs and transesophageal echocardiography findings, raising suspicion for VAE. Immediate management, including surgical field protection and cardiopulmonary support, stabilized the patients’ conditions. The procedure was subsequently discontinued in case 1 and modified to limited resection in case 2. Postoperative computed tomography revealed intracranial venous air within the internal jugular vein, cavernous sinus, and diploic veins.<br>
Conclusion: These cases highlight that VAE can occur even before dural incision. Vigilant intraoperative monitoring and prompt intervention are essential for preventing potentially fatal outcomes.No potential conflict of interest relevant to this article was reported.The Korean Society of AnesthesiologistsActa Medica Okayama2005-64197752024Utilization of the pericapsular nerve group block in preoperative rehabilitation of patients with femoral neck fractures -a case series-565569ENZhuanJinDepartment of Anesthesiology and Resuscitology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesDaisukeSugiyamaDepartment of Anesthesiology, Kameda Medical CenterFumiyaHigoDepartment of Rehabilitation, Kameda Medical CenterTakahiroHirataDepartment of Rehabilitation, Kameda Medical CenterOsamuKobayashiDepartment of Anesthesiology, Kameda Medical CenterHiroshiMorimatsuDepartment of Anesthesiology and Resuscitology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenichiUedaDepartment of Anesthesiology, Kameda Medical CenterBackground: Elderly patients with femoral neck fractures, particularly those with severe comorbidities or living in regions with limited medical resources, may experience delays in surgical treatment. Although the benefits of preoperative rehabilitation (prehabilitation) in hip arthroplasty have been reported, pain management remains a challenge. The pericapsular nerve group (PENG) block, known for its exceptional analgesic effect and motor function preservation, may be a promising intervention during prehabilitation in these patients.<br>
Case: We enrolled ten patients with Garden classification 3–4 femoral neck fractures scheduled for hip arthroplasty. After receiving a PENG block with 20 ml of 0.375% ropivacaine, all patients underwent initial prehabilitation sessions comprising 9 mobility levels, ranging from bed-sitting to walking. One patient was excluded due to experiencing high blood pressure during prehabilitation. Six of the nine remaining patients (66.7%) were successfully transferred from bed to wheelchair.<br>
Conclusions: The PENG block enhanced prehabilitation for patients with femoral neck fractures undergoing hip arthroplasty.No potential conflict of interest relevant to this article was reported.Journal of Neurosurgery Publishing Group (JNSPG)Acta Medica Okayama2694-190210132025Endovascular treatment for a symptomatic dissecting ophthalmic artery aneurysm occurring in the orbit: illustrative caseCASE25483ENKoheiIzumiharaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunHarumaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenjiSugiuDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFukikoBabaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJuntaroFujitaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYuichiHirataDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYutaSotomeDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasatoKawakamiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyuKimuraDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasafumiHiramatsuDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShotaTanakaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesBACKGROUND: Peripheral ophthalmic artery aneurysms (POAAs) arising from the main trunk or branches of the ophthalmic artery (OphA) are extremely rare. However, their epidemiology and optimal management remain poorly understood. The authors report a rare case of a symptomatic POAA caused by arterial dissection that was successfully treated using endovascular therapy, leading to favorable visual recovery.<br>
OBSERVATIONS: A 77-year-old woman presented with sudden-onset visual impairment in the right eye. Ophthalmological examination revealed a defect in the right visual field. CT angiography revealed a fusiform aneurysm in the right intraorbital OphA. Digital subtraction angiography revealed a pearl and string sign, consistent with a dissecting aneurysm. A balloon test occlusion (BTO) of the OphA origin confirmed collateral circulation from the external carotid artery. Internal trapping of the OphA was performed under general anesthesia. Postoperatively, the patient’s visual function gradually improved, and complete recovery was achieved within 3 months.<br>
LESSONS: Although POAAs are exceptionally rare, they may lead to significant visual dysfunction owing to optic nerve compression. When visual symptoms are present, prompt intervention may reverse the symptoms. Preoperative assessment of collateral circulation using BTO is essential for treatment planning. Internal trapping may be an effective strategy when sufficient collateral flow is confirmed.No potential conflict of interest relevant to this article was reported.Japan Neurosurgical SocietyActa Medica Okayama2188-4226122025Endovascular Thrombectomy for Large Vessel Occlusion in a Patient on Venoarterial Extracorporeal Membrane Oxygenation: A Case Report445451ENYukiEBISUDANIDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasafumiHIRAMATSUDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKeiichiroIWASAKIDepartment of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenjiSUGIUDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunHARUMADepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyuKIMURADepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasatoKAWAKAMIDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYutaSOTOMEDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakahiroNISHIHARADepartment of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinsukeYUASADepartment of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShotaTANAKADepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesExtracorporeal membrane oxygenation is utilized in the treatment of severe acute cardiac failure and respiratory failure. While it provides the advantage of oxygenating blood through extracorporeal circulation, it also carries risks of intracranial ischemic and hemorrhagic complications due to the continuous presence of artificial materials within the body. We encountered a case in which venoarterial extracorporeal membrane oxygenation was initiated for fulminant myocarditis, and the patient subsequently developed a large vessel occlusion. The diagnosis was confirmed using perfusion computed tomography. A visible thrombus was observed on the arterial cannula of the extracorporeal membrane oxygenation circuit, and the large vessel occlusion was determined to have been caused by thromboembolism. An immediate extracorporeal membrane oxygenation circuit exchange was performed, followed by endovascular thrombectomy. The patient experienced no perioperative complications and achieved a favorable neurological outcome. Endovascular thrombectomy in extracorporeal membrane oxygenation patients requires careful perioperative management and should be promptly performed in eligible cases of thromboembolic events. Furthermore, because patients on extracorporeal membrane oxygenation are often sedated and under intensive systemic management, regular neurological assessments and intracranial monitoring are essential for the early detection of intracranial pathologies.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama0385-240752102025Biologics and Small‐Molecule Therapies in Netherton Syndrome: A Comprehensive Review14831493ENShinMorizaneDepartment of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomoyukiMukaiDepartment of Immunology and Molecular Genetics, Kawasaki Medical SchoolKoSunagawaDepartment of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKen‐ichiHasuiDepartment of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAnriMoritaDepartment of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHayatoNomuraDepartment of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMamoruOuchidaDepartment of Molecular Oncology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesNetherton syndrome (NS) is a rare congenital ichthyosis caused by loss-of-function mutations in the SPINK5 gene, leading to defective expression of the serine protease inhibitor LEKTI. Dysregulated epidermal protease activity results in impaired skin barrier function and chronic inflammation, accompanied by complex immune profiles. NS patients commonly show activation of the inflammatory axis, centered on IL-17 and IL-36, in the skin and blood, and show a psoriasis-like shift to Th17. Conversely, the immune profile differs depending on the clinical type, with ichthyosis linearis circumflexa type characterized by complement activation and Th2-type allergic responses, and scaly erythroderma type characterized by a type I IFN signature and Th9-type allergic responses. While symptomatic treatments such as emollients and topical corticosteroids have been the mainstay of care, recent advances have opened new therapeutic avenues involving biologic agents and oral small-molecule immunomodulators. This review provides a comprehensive overview of the current clinical landscape and future directions of biologics (e.g., dupilumab, secukinumab, ustekinumab) and small-molecule therapies (e.g., JAK inhibitors such as tofacitinib, baricitinib, and upadacitinib) in the treatment of NS. Though evidence remains limited to case reports and small series, preliminary data suggest that cytokine-targeted interventions―particularly those inhibiting IL-4, IL-13, IL-17, IL-36, and JAK pathways―may offer tangible clinical benefits. Well-designed clinical trials and mechanistic investigations are crucial to establishing their place in NS management.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-818417102025Histopathological Study of Regenerative Endodontic Therapy on an Immature Mandibular Second Premolar With Pulp Necrosis: A Case Reporte95647ENHidefumiSakoDepartment of Periodontics and Endodontics, Division of Dentistry, Okayama University HospitalKazuhiroOmoriDepartment of Pathophysiology - Periodontal Science, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYukiShinoda-ItoDepartment of Pathophysiology - Periodontal Science, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKiyofumiTakabatakeDepartment of Oral Pathology, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityHitoshiNagatsukaDepartment of Oral Pathology, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityShogoTakashibaDepartment of Pathophysiology - Periodontal Science, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityRegenerative endodontic therapy (revascularization) for immature permanent teeth with pulp necrosis and/or apical periodontitis is an effective treatment to promote root maturation. Previous histological studies have reported the formation of cementoid or osteoid tissue and periodontal ligament-like tissue within the root canals. This case report presents the histopathological findings of a human immature permanent tooth with pulp necrosis following revascularization.<br>
<br>
A 11-year-old male patient presented with tenderness on biting and the formation of a sinus tract in the mandibular right second premolar (tooth #29), diagnosed as pulp necrosis with symptomatic apical periodontitis. Revascularization was performed using calcium hydroxide as an intracanal medicament, with reference to the American Association of Endodontists (AAE) 2018 Position Paper on Regenerative Endodontics. At the 12-month follow-up, radiographs showed thickening of the canal walls, apical narrowing, root elongation, and recovery of pulp sensibility. The tooth was later extracted for orthodontic reasons at 42 months and processed for histological examination.<br>
<br>
Histological evaluation revealed cementum-like hard tissue continuous with the existing dentin in the apical region, suggesting apical closure. In contrast, the coronal portion showed less mature cementum-like tissue accompanied by loose connective tissue and neovascularization. These findings indicate that revascularization with calcium hydroxide can induce the formation of cementum-like and dentin-like tissues with vascular regeneration in immature permanent teeth with pulp necrosis.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama1086-93792025The effect of pressure on dihedral angle between liquid Fe‐S and orthopyroxene: Implication for percolative core formation in planetesimals and planetary embryosENTakumiMiuraDepartment of Earth and Space Science, Osaka UniversityHidenoriTerasakiDepartment of Earth Sciences, Okayama UniversityHyuTakakiDepartment of Earth Sciences, Okayama UniversityKotaroKobayashiDepartment of Earth Sciences, Okayama UniversityGeoffrey DavidBromileySchool of Geosciences, The University of EdinburghTakashiYoshinoInstitute for Planetary Materials, Okayama UniversityDuring precursor stages of planet formation, many planetesimals and planetary embryos are considered to have differentiated, forming an iron-alloy core and silicate mantle. Percolation of liquid iron-alloy in solid silicates is one of the major possible differentiation processes in these small bodies. Based on the dihedral angles between Fe-S melts and olivine, a criterion for determining whether melt can percolate through a solid, it has been reported that Fe-S melt can percolate through olivine matrices below 3 GPa in an oxidized environment. However, the dihedral angle between Fe-S melts and orthopyroxene (opx), the second most abundant mineral in the mantles of small bodies, has not yet been determined. In this study, high-pressure and high-temperature experiments were conducted under the conditions of planetesimal and planetary embryo interiors, 0.5–5.0 GPa, to determine the effect of pressure on the dihedral angle between Fe-S melts and opx. Dihedral angles tend to increase with pressure, although the pressure dependence is markedly reduced above 4 GPa. The dihedral angle is below the percolation threshold of 60° at pressures below 1.0–1.5 GPa, indicating that percolative core formation is possible in opx-rich interiors of bodies where internal pressures are lower than 1.0–1.5 GPa. The oxygen content of Fe-S melt decreases with increasing pressure. High oxygen contents in Fe-S melt reduce interfacial tension between Fe-S melt and opx, resulting in reduced dihedral angles at low pressure. Combined with previous results for dihedral angle variation of the olivine/Fe-S system, percolative core formation possibly occurs throughout bodies up to a radius of 1340 km for an olivine-dominated mantle, and up to 770 km for an opx-dominated mantle, in the case of S-rich cores segregating under relatively oxidizing conditions. For mantles of small bodies in which abundant olivine and opx coexist, the mineral with the largest volume fraction and/or smallest grain size will allow formation of interconnected mineral channels, and, therefore, the wetting property of this mineral determines the wettability of the melt, that is, controls core formation.No potential conflict of interest relevant to this article was reported.Ovid Technologies (Wolters Kluwer Health)Acta Medica Okayama2376-78391152025Vanishing White Matter Disease With EIF2B2 c.254T >A Variante200293ENToshiyukiKakumotoDepartment of Neurology, Graduate School of Medicine, The University of TokyoTakashiMatsukawaDepartment of Neurology, Graduate School of Medicine, The University of TokyoRyoTokimuraDepartment of Neurology, Graduate School of Medicine, The University of TokyoYokoTsuboyamaDepartment of Neurology, Graduate School of Medicine, The University of TokyoYasufumiHayashiDepartment of Neurology, Graduate School of Medicine, The University of TokyoAkihikoMitsutakeDepartment of Neurology, Graduate School of Medicine, The University of TokyoAtsushiIwataDepartment of Neurology, Graduate School of Medicine, The University of TokyoMeiko HashimotoMaedaDepartment of Neurology, Graduate School of Medicine, The University of TokyoJunShimizuDepartment of Neurology, Graduate School of Medicine, The University of TokyoWataruGonoiDepartment of Radiology, Graduate School of Medicine, The University of TokyoHiroyukiIshiuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunMitsuiDepartment of Neurology, Graduate School of Medicine, The University of TokyoShojiTsujiDepartment of Molecular Neurology, Graduate School of Medicine, The University of TokyoTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of TokyoObjectives<br>
Typical MRI findings of vanishing white matter disease (VWM) include diffuse white matter lesions with cystic degeneration. However, mild cases may lack these typical features, posing diagnostic challenges.<br>
Methods<br>
We describe 2 of 3 individuals carrying the homozygous c.254T >A variant in EIF2B2 identified at our hospital, excluding 1 previously reported case.1 Genetic analyses were performed using whole-genome sequence or whole-exome sequence analysis, and detected variants were confirmed by direct nucleotide sequence analysis. Brain MRI findings and clinical features were reviewed for the 2 individuals along with other cases in the literature with the same variant.<br>
Results<br>
A 69-year-old woman presented with recurrent transient dizziness and secondary amenorrhea. MRI of the brain revealed small T2-hyperintense lesions confined to the subcortical white matter with hyperintensities on diffusion-weighted images and mildly elevated apparent diffusion coefficient values. A 28-year-old woman presented with transient dizziness and secondary amenorrhea. MRI of the brain showed mild T2-hyperintense lesions in the cerebral white matter with frontal predominance.<br>
Discussion<br>
This report highlights the clinically mild cases of VWM with subtle abnormalities on brain MRI who had the homozygous c.254T >A in EIF2B2, further expanding the clinical spectrum of VWM and underscoring the importance of genetic assessments in the diagnosis of individuals with mild clinical and MRI findings.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841782025Subacute Progression of Gait Disturbance and Consciousness Impairment Due to Communicating Hydrocephalus Associated With Vestibular Schwannomae89880ENSatokaYanoDepartment of Neurology, The University of Tokyo Graduate School of Medicine and Faculty of MedicineAkatsukiKubotaDepartment of Neurology, The University of Tokyo Graduate School of Medicine and Faculty of MedicineMizuhoKawaiDepartment of Neurology, The University of Tokyo Graduate School of Medicine and Faculty of MedicineDaikiYashitaDepartment of Neurology, The University of Tokyo Graduate School of Medicine and Faculty of MedicineHiroyukiIshiuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesWataruSatakeDepartment of Neurology, The University of Tokyo Graduate School of Medicine and Faculty of MedicineKaoruYamadaDepartment of Neuropathology, The University of Tokyo Graduate School of Medicine and Faculty of MedicineYukiShinyaDepartment of Neurosurgery, The University of Tokyo Graduate School of Medicine and Faculty of MedicineSatoruMiyawakiDepartment of Neurosurgery, The University of Tokyo Graduate School of Medicine and Faculty of MedicineTakeshiIwatsuboDepartment of Neuropathology, The University of Tokyo Graduate School of Medicine and Faculty of MedicineTatsushiTodaDepartment of Neurology, The University of Tokyo Graduate School of Medicine and Faculty of MedicinePatients with vestibular schwannomas (VSs) present with vestibulocochlear nerve dysfunction such as vertigo and tinnitus. VSs occasionally develop communicating hydrocephalus as a complication, which is typically characterized by an insidious progression of symptoms. We report a case of an 84-year-old female patient with a VS who developed gait disturbance and consciousness impairment over a three-week period, ultimately resulting in an inability to walk and communicate. A thorough evaluation ruled out encephalitis and other differential diagnoses. Imaging studies demonstrated findings consistent with communicating hydrocephalus, and a tap test temporarily improved her consciousness disturbances. The patient underwent ventriculoperitoneal shunting and stereotactic radiosurgery (SRS), after which both consciousness and gait disturbances dramatically improved 10 days postoperatively. The subacute development of symptoms due to normal pressure hydrocephalus associated with VSs is rare. Furthermore, to the best of our knowledge, this is the first reported case of severe gait impairment and disturbance of consciousness progressing within a short period. This case highlights the importance of considering communicating hydrocephalus associated with VSs as a differential diagnosis, even in cases of subacute consciousness disturbance. We also discuss the pathophysiology of hydrocephalus in relation to cerebrospinal fluid (CSF) clearance into the extracranial space. No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1752-19471912025The safety and efficacy of finasteride for transgender men with androgenetic alopecia: a case series468ENYusukeTominagaDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineTomokoKobayashiDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYukoMatsumotoDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineTomokoSakoDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalTakatoshiMoriwakeDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineSatoshiHoriiDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineTakuyaSadahiraDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineSatoshiKatayamaDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineTakehiroIwataDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShingoNishimuraDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKensukeBekkuDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKoheiEdamuraDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineMasamiWatanabeCenter for Innovative Clinical Medicine, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineMotooArakiDepartment of Urology, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineBackground Testosterone replacement therapy is commonly used in transgender men for masculinization. One of the most common adverse effects of testosterone replacement therapy is androgenetic alopecia. In Japan, finasteride is approved exclusively for cisgender men and is not indicated for transgender men. The aim of this clinical trial was to evaluate the safety and efficacy of finasteride in transgender men with androgenetic alopecia.<br>
Case presentation This study included three transgender men (assigned female at birth, identifying as male), aged 44, 43, and 29 years. All participants were of Asian ethnicity. A clinical trial was conducted from October 2021 to December 2023. Transgender men aged 20–60 years who had not undergone hysterectomy, were undergoing testosterone replacement therapy, and who had been diagnosed with stage ≥ II androgenetic alopecia on the basis of the Norwood–Hamilton scale were recruited. The participants initiated treatment with 0.2 mg of finasteride per day for 3 months (phase 1). If no adverse events above grade 2 occurred, the dose was increased to 1.0 mg per day for an additional 3 months (phase 2). The primary endpoints were the incidence of treatment-related adverse events at 1 week, 1 month, and 3 months, as well as the rate of participants continuing treatment at 3 months. None of the patients experienced serious adverse events at 3 months, and all the patients extended their treatment to a total of 6 months. Improvements of at least one stage on the N–H scale were observed, but two participants experienced resumption of menstruation.<br>
Conclusion Finasteride appears to be a safe and effective treatment for androgenetic alopecia in transgender men undergoing testosterone replacement therapy. However, its potential for reducing some of the effects of testosterone replacement therapy warrants further investigation. Trial registration: jRCT, jRCTs061210040, registered 7 October 2021, https://jrct.mhlw.go.jp/latest-detail/jRCTs061210040.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7952025Epstein-Barr Virus-Associated Early Gastric Carcinoma with Lymphoid Stroma Mimicking a Submucosal Tumor: A Typical Case Diagnosed by Endoscopic Resection and Treated by Local Resection with Sentinel Node Navigation399404ENHiroshiIsozakiDepartment of Surgery, Oomoto HospitalSasauMatsumotoDepartment of Surgery, Oomoto HospitalTakehiroTakamaDepartment of Surgery, Oomoto HospitalYukaIsozakiDepartment of Surgery, Oomoto HospitalShigekiMurakamiDepartment of Surgery, Oomoto HospitalCase Report10.18926/AMO/69442Gastric cancer with lymphoid stroma (GCLS) accounts for 1%-7% of gastric cancers; ~80% are Epstein-Barr virus (EBV)-positive. The rate of lymph node metastasis is relatively low, even when an early GCLS has invaded the submucosa. We report an early GCLS with massive submucosal invasion mimicking a submucosal tumor (SMT), diagnosed by endoscopic submucosal resection (ESD) and treated with local resection and sentinel node navigation surgery (SNNS). The patient was a 40-year-old Japanese man. A protruding lesion on the greater curvature of the middle part of his stomach was detected by X-ray, and an endoscopic examination revealed a 2.5-cm protruding tumor covered with a normal mucosa and small ulcers at the apex. ESD was performed for a diagnosis. The pathological diagnosis was lymphoepithelioma-like gastric cancer (GCLS), pT1b(SM2), Ly0, V0, pHM1, pVM1. EBV infection in the cancer cells was confirmed pathologically by EBV-encoded RNA. The local resection was performed using SNNS. The patient has had no recurrence or post-gastrectomy syndrome 4 years postsurgery. EBV-associated early GCLS resembling an SMT is relatively rare, and clinicians need to be aware of this disease. Local resection using SNNS may be a surgical option for GCLS cases with a low rate of lymphatic metastasis.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7952025Gastroduodenal Artery-Preserving Pancreatoduodenectomy after Esophagectomy with Gastric Conduit Reconstruction393398ENAkariMasunagaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKoseiTakagiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYasuoNagaiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazuyaYasuiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomokazuFujiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMotohikoYamadaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakeyoshiNishiyamaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesNoriyukiKanehiraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYumiSotaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/69441Pancreatoduodenectomy (PD) after esophagectomy with gastric conduit reconstruction is technically challenging. Preserving the blood supply of the gastric conduit is crucial in performing PD after esophagectomy. We report the case of a 66-year-old man who underwent gastroduodenal artery-preserving PD after esophagectomy with gastric conduit reconstruction for intraductal papillary mucinous neoplasm. The patient developed pseudoaneurysm rupture postoperatively, but was successfully treated with interventional radiology. Precise assessment is important in developing a surgical strategy depending on the patient’s specific anatomy and tumor characteristics. Moreover, special attention should be paid to avoid accidental injuries of the gastric conduit and gastric vessels during surgery.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7952025The Utility of a Preoperative 3D Imaging Analysis System for Trigonal Meningioma387392ENYusukeMoriDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshihiroOtaniDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyoOmaeDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShuichiroHiranoDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJojiIshidaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKentaroFujiiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunHarumaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasafumiHiramatsuDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesToshiMatsushitaDivision of Radiological Technology, Okayama University HospitalFumiyoHigakiDepartment of Radiology, Medical Development Field, Okayama UniversityKenjiSugiuDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShotaTanakaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/69440Trigonal meningiomas are rare and pose surgical challenges due to their deep location and proximity to critical neuroanatomical structures. We present the case of a 67-year-old woman with a growing trigonal meningioma successfully resected with guidance by a preoperative 3D imaging analysis system. Integration of CT and MRI including diffusion tensor imaging (DTI) enabled precise mapping of the optic radiation, guiding a middle temporal gyrus approach. Preoperative embolization reduced tumor vascularity, facilitating gross total resection with minimal blood loss. This case highlights the effectiveness of preoperative 3D imaging systems in optimizing surgical planning and improving outcomes in complex neurosurgical cases.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7952025Immunoglobulin G4-related Disease Mimicking Portal Vein Tumor Thrombus381385ENAtsunobuSakuraiDepartment of Radiology, NHO Iwakuni Clinical CenterTakayukiYabukiDepartment of Radiology, NHO Iwakuni Clinical CenterHidekiAokiDepartment of Gastroenterological Surgery, NHO Iwakuni Clinical CenterAkikoIsekiDepartment of Pathology, NHO Iwakuni Clinical CenterCase Report10.18926/AMO/69439We report the case of a 72-year-old Japanese man with an incidental portal vein mass that was surgically resected and diagnosed as immunoglobulin G4 (IgG4)-related disease. The mass was discovered during an atrial fibrillation examination. The patient had a history of gastric cancer and was also diagnosed with rectal cancer, raising concerns about metastasis. Due to technical challenges, a biopsy was not feasible. Imaging findings suggested portal vein tumor thrombosis, complicating the diagnosis. This case highlights a rare presentation of IgG4-related disease mimicking portal vein tumor thrombus.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7952025A Review of the Endoscopic Treatment for Bile Leak Following Cholecystectomy and Hepatic Surgery321328ENTaisukeObataDepartment of Gastroenterology and Hepatology, Okayama University HospitalKazuyukiMatsumotoDepartment of Gastroenterology and Hepatology, Okayama University HospitalMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University HospitalReview10.18926/AMO/69432Bile leak occurs in 2-25% of liver transplant, 3-27% of hepatic resection, and 0.1-4% of cholecystectomy cases. The clinical course of bile leak varies depending on the type of surgery that caused the fistula, as well as the type, severity, and timing of bile duct injury. Although infections resulting from bile leak can be life-threatening, the introduction of endoscopic treatment has enabled some patients to avoid reoperation and has reduced the negative impact on quality of life associated with external fistulas for percutaneous drainage. Endoscopic interventions, such as sphincterotomy and stent placement, reduce the pressure gradient between the bile duct and duodenum, facilitating bile drainage through the papilla and promoting the closure of the leak. We reviewed the literature from 2004 to 2024 regarding bile leak following cholecystectomy and liver surgery, examining recommended techniques, timing, and treatment outcomes. In cases of bile leak following cholecystectomy, clinical success was achieved in 72-96% of cases, while success rates for bile leak following liver surgery ranged from 50% to 100%. Although endoscopic treatment is effective, it is not universally applicable, and its limitations must be carefully considered.No potential conflict of interest relevant to this article was reported.Japanese Society of Internal MedicineActa Medica Okayama0918-291864202025Two Cases of Esophageal Mucosal Damage Observed after Peroral Endoscopic Myotomy for Esophageal Motility Disorders29792984ENShoichiroHirataDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomohiroKamioDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTakuyaSatomiDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKentaHamadaDepartment of Practical Gastrointestinal Endoscopy, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesHiroyukiSakaeDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesSeijiKawanoDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYoshiroKawaharaDepartment of Practical Gastrointestinal Endoscopy, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesNoriakiManabeDivision of Endoscopy and Ultrasonography, Department of Clinical Pathology and Laboratory Medicine, Kawasaki Medical SchoolMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesThis report presents two cases of esophageal mucosal damage following peroral endoscopic myotomy (POEM) for esophageal motility disorders. In the first case, delayed perforation and mediastinitis occurred on postoperative day 15 and the patient was treated with endoscopic clipping and antibiotics. In the second case, although no perforation was observed, extensive mucosal injury developed the day after POEM which was successfully managed by fasting and antibiotic therapy. These findings highlight the need for careful patient management to minimize the risks associated with POEM, while maximizing its therapeutic benefits.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1865-72572025Metachronic development of cholangiocarcinoma during treatment for IgG4-related sclerosing cholangitisENKosakuMorimotoDepartment of Internal Medicine, Tsuyama Chuo HospitalKazuyukiMatsumotoDepartment of Gastroenterology and Hepatology, Okayama University HospitalTakakiOkuyamaDepartment of Internal Medicine, Tsuyama Chuo HospitalShogoKimuraDepartment of Internal Medicine, Tsuyama Chuo HospitalKensukeTakeiDepartment of Internal Medicine, Tsuyama Chuo HospitalTakuyaSatomiDepartment of Internal Medicine, Tsuyama Chuo HospitalTsuyoshiOkadaDepartment of Surgery, Tsuyama Chuo HospitalSusumuShinouraDepartment of Surgery, Tsuyama Chuo HospitalReiShibataDepartment of Diagnostic Pathology, Tsuyama Chuo HospitalRyutaTakenakaDepartment of Internal Medicine, Tsuyama Chuo HospitalWe report a case of obstructive jaundice due to recurrent distal biliary stricture during 3 years of treatment for immunoglobulin G4 (IgG4)-related sclerosing cholangitis (IgG4-SC) associated with autoimmune pancreatitis. Although a relapse of IgG4-SC was initially suspected, imaging findings, laboratory tests, and histopathological examinations led to the diagnosis of metachronous cholangiocarcinoma. The patient underwent pancreaticoduodenectomy, and no cancer recurrence was noted 6 months postoperatively. Distal cholangiocarcinoma and IgG4-SC remission were observed in the resected specimen. In patients with recurrent biliary strictures during IgG4-SC treatment, comprehensive evaluations are essential because of the risk of disease relapse and development of metachronous cholangiocarcinoma.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2045-23221512025Lactose fermenting enteroinvasive Escherichia coli from diarrhoeal cases confers enhanced virulence24040ENDebjaniGhoshDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)ProlayHalderDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)ProsenjitSamantaDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)GoutamChowdhuryCollaborative Research Centre of Okayama University for Infectious Diseases, ICMR-National Institute for Research in Bacterial InfectionsSreejaShawDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)PujaBoseDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)DeboleenaRoyDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)NiveditaRoyDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)KeiKitaharaCollaborative Research Centre of Okayama University for Infectious Diseases, ICMR-National Institute for Research in Bacterial InfectionsThandavarayanRamamurthyDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)HemantaKoleyDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)Shin-ichiMiyoshiGraduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityShantaDuttaDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)Asish KumarMukhopadhyayDivision of Bacteriology, ICMR-National Institute for Research in Bacterial Infections (ICMR-NIRBI)Enteroinvasive Escherichia coli (EIEC), known for causing bacillary dysentery akin to Shigella species, comprises both lactose-fermenting (LF) and non-lactose-fermenting (NLF) isolates. While NLF-EIEC is a well-established pathogen associated with acute dysentery and harbours classical Shigella-like virulence factors, the role of LF-EIEC in human disease remains underexplored. In this study, we sought to characterize LF-EIEC clinical isolates and assessed their pathogenic potential in comparison to NLF-EIEC. Among 13,682 diarrhoeal stool specimens, six LF and nine NLF-EIEC were isolated, predominantly belonging to serogroups O28ac, O125, O136, and O152. Unlike other E. coli, all the EIEC isolates were non-motile. Both the types of EIEC had multiple plasmids harbouring several virulence encoding genes (ipaBCD, ial, virF, sig, sepA and ipaH). Resistance to recent generation antibiotics were mostly confined to NLF-EIEC but some of the LF-EIEC were resistant only to ceftriaxone. Higher invasion ability and significant increase in the expression of virulence encoding genes by the LF-EIEC (p < 0.05) were noted during infection to Int407 cell-line. Additionally, LF-EIEC exhibited extensive colonization of the mouse intestine and expressed severe keratoconjunctivitis in guinea pigs. Together, our findings highlight LF-EIEC as an emerging pathogenic variant warranting heightened surveillance and comprehensive investigation to better understand its epidemiological and clinical significance.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2692-4609612025Adequacy evaluation of 22‐gauge needle endoscopic ultrasound‐guided tissue acquisition samples and glass slides preparation for successful comprehensive genomic profiling testing: A single institute experiencee70104ENTamiNagataniClinical Genomic Medicine, Dentistry and Pharmaceutical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYojiWaniDepartment of Pathology, Japanese Red Cross Society, Himeji Red Cross HospitalMasahiroTakataniDepartment of Internal Medicine, Japanese Red Cross Society, Himeji Red Cross HospitalSoichiroFushimiDepartment of Pathology, Japanese Red Cross Society, Himeji Red Cross HospitalHirofumiInoueDivision of Medical Support, Dentistry and Pharmaceutical Science, Okayama University Graduate School of MedicineShinichiroHoriDepartment of Internal Medicine, Japanese Red Cross Society, Himeji Red Cross HospitalKyoheiKaiDepartment of Genetic Medicine, Japanese Red Cross Society, Himeji Red Cross HospitalHidekiYamamotoClinical Genomic Medicine, Dentistry and Pharmaceutical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTetsuyaOkazakiClinical Genomic Medicine, Dentistry and Pharmaceutical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMakiTaniokaClinical Genomic Medicine, Dentistry and Pharmaceutical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHiroyukiOkadaDepartment of Internal Medicine, Japanese Red Cross Society, Himeji Red Cross HospitalAkiraHirasawaClinical Genomic Medicine, Dentistry and Pharmaceutical Science, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesObjectives: This study aimed to evaluate the successful sequencing rate of Foundation One CDx (F1CDx) using small tissue samples obtained with a 22-gauge needle (22G) through endoscopic ultrasound-guided fine needle acquisition (EUS-TA) and to propose guidelines for tissue quantity evaluation criteria and proper slide preparation in clinical practice.<br>
Methods: Between June 2019 and April 2024, 119 samples of 22G EUS-TA collected for F1CDx testing at Himeji Red Cross Hospital were retrospectively reviewed. Tissue adequacy was only assessed based on tumor cell percentage (≥20%). The procedure stopped when white tissue fragments reached 20 mm during macroscopic on-site evaluation. The specimens were prepared using both ‘tissue preserving sectioning’ to retain tissue within formalin-fixed paraffin-embedded blocks and the ‘thin sectioning matched needle gauge and tissue length’ method with calculation to ensure minimal unstained slides for the 1 mm3 sample volume criterion. Tissue area from HE slides and sample volume were measured, and F1CDx reports were analyzed.<br>
Results: Of 119 samples, 108 (90.8%) were suitable for F1CDx. Excluding the cases not submitted for testing, in the 45 cases where F1CDx was done using 22G EUS-TA samples, eight (17.8%) had a sum of tissue area tissue of 25 mm2 or greater in the HE-stained sample. However, all cases met the F1CDx 1 mm3 volume criterion by submitting > 30 unstained slides per sample. As a result, 43 of 45 cases (95.6%) were successfully analyzable.<br>
Conclusions: The 22G EUS-TA needle is an effective tool for providing the sufficient tissue volume required for F1CDx.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841782025Craniofacial Fibrous Dysplasia to Affect or Not the Optic Nerve in Long-Term Follow-Up of Three Casese91072ENToshihikoMatsuoDepartment of Ophthalmology, Graduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama University, Okayama University HospitalTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityKiyoshiYamadaDepartment of Plastic and Reconstructive Surgery, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityMitsuhiroOkanoDepartment of Otorhinolaryngology, School of Medicine, International University of Health and WelfareFibrous dysplasia of the bone is characterized by immature fibrous bones of trabeculae and fibrovascular proliferation in the medulla. In this study, we report three consecutive patients with craniofacial fibrous dysplasia with or without optic nerve involvement. In Case 1, a 43-year-old man with blurred vision in the right eye at the first visit was well until the age of 54 years, when he came back with symptoms suggestive of paranasal sinusitis. Computed tomography scans disclosed a mucocele in the right sphenoid sinus and thickened bilateral ethmoid, sphenoid, and frontal bones. He underwent an emergency nasal endoscopic surgery to make a drainage opening to the sphenoid and ethmoid sinuses on the right side with incomplete success. The pathology of the resected tissue confirmed fibrous dysplasia. With intravenous antibiotics, he recovered from blepharoptosis, complete ophthalmoplegia, and visual acuity decrease on the right side. He was well until the age of 71 years when he had a self-limiting episode of visual field cloudiness caused by the right sphenoid sinus mucocele. At the age of 75 years, he developed abrupt vision loss to no light perception in the right eye. He underwent an open skull surgery to extirpate the sphenoid mucocele on the right side and died of an unknown cause two years later. In Case 2, a 29-year-old man had a two-week-long headache, and computed tomography scans revealed fibrous dysplasia in the bilateral sphenoid bones. Nasal biopsy at the spheno-ethmoid recess proved a pathological diagnosis of fibrous dysplasia. Goldmann perimetry showed normal visual fields in both eyes. He was followed every year by magnetic resonance imaging to maintain normal visual fields until the latest visit at the age of 41 years. In Case 3, a 12-year-old girl was referred to an ophthalmologist to check her vision. She had been diagnosed with fibrous dysplasia of the left maxillary bone at the age of six years by a dentist. She had a gingival resection on the left maxilla at the age of 15 years and had a left maxillary bone resection at 18 years at another hospital. One month after the resection, Goldmann perimetry showed superior peripheral field depression in the left eye, in contrast with the normal visual field in the right eye. She maintained the visual acuity of 1.5 in both eyes until the last visit at the age of 21 years. In fibrous dysplasia as a rare disease, functional and cosmetic problems, including vision problems, should be considered in a case-based approach.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2769-2558432025Occupational therapist‐guided exercise increased white blood cell and neutrophil counts during clozapine treatment: A case reporte70167ENKenjiHinotsuDepartment of Neuropsychiatry, Okayama University HospitalShinjiSakamotoDepartment of Neuropsychiatry, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesHirokiKawaiDepartment of Neuropsychiatry, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshioOhyaDepartment of Neuropsychiatry, Okayama University HospitalAkiyoshiYokodeDepartment of Neuropsychiatry, Okayama University HospitalTakahiroAsadaDepartment of Neuropsychiatry, Okayama University HospitalYukoOkahisaDepartment of Neuropsychiatry, Okayama University HospitalManabuTakakiDepartment of Neuropsychiatry, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesBackground: Moderate exercise increases white blood cells and neutrophils. However, there are no reports on the relationship between exercise intensity and these cells. We observed a patient taking clozapine whose white blood cell and neutrophil counts were borderline. Supervised exercise therapy with an occupational therapist stabilized these counts.<Br>
Case Presentation: A 50-year-old woman with treatment-resistant schizophrenia was prescribed clozapine. By Day 63, the clozapine dosage had been increased to 450 mg/day. Additionally, she was advised to perform a 30-min walking exercise program 1 h before blood tests. Exercise therapy supervised by an occupational therapist was performed eight times, and self-training was performed five times. Exercise intensity was monitored using the Borg Scale for subjective evaluation and the Karvonen formula for objective evaluation. Supervised exercise therapy with an occupational therapist resulted in greater increases on the Borg Scale and Karvonen formula than did self-training. It also induced increases in white blood cells and neutrophils. Her psychiatric symptoms improved, and she was discharged on Day 71. A blood test taken after discharge revealed that her white blood cell and neutrophil counts were within the normal range and she continued to take clozapine for 2 years. She has since been able to enjoy a calm and relaxed life at home.<br>
Conclusion: Exercise involving subjective and objective evaluation by an occupational therapist effectively increased white blood cells and neutrophils during clozapine treatment. Supervised exercise therapy by an occupational therapist is important when self-exercise is insufficient for continuing clozapine treatment.No potential conflict of interest relevant to this article was reported.Japan Surgical SocietyActa Medica Okayama2198-77931112025Obese Patient with Gastric Diverticulum Undergoing Laparoscopic Sleeve Gastrectomy Guided by Preoperative Endoscopic Measurement: A Case Report and Literature Reviewcr.25-0141ENKensukeHirosunaCenter for Graduate Medical Education, Okayama University HospitalHajimeKashimaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesRyoheiShojiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYukiMatsumiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYoshihikoKakiuchiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesSatoruKikuchiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesShinjiKurodaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesFuminoriTeraishiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesShunsukeKagawaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesINTRODUCTION: Gastric diverticulum is a rare condition, often asymptomatic and incidentally detected. Laparoscopic sleeve gastrectomy (LSG) is a widely performed bariatric procedure, but a gastric diverticulum complicates surgical planning. In this case, careful preoperative assessment allowed safe execution of LSG despite the diverticulum’s proximity to the esophagogastric junction.<br>
CASE PRESENTATION: A 45-year-old woman (BMI: 46.8 kg/m2) with hypertension, dyslipidemia, and glucose intolerance was referred for bariatric surgery after unsuccessful weight loss with conservative management. Preoperative endoscopy revealed an 18 × 14 mm gastric diverticulum on the posterior wall of the gastric fundus, 40 mm from the esophagogastric junction. LSG was performed using a surgical stapler, ensuring complete diverticulum resection while preserving gastric tube integrity. The surgery was uneventful, with minimal blood loss and a duration of 2 hours and 52 minutes. The patient had an uneventful postoperative course and was discharged on day 9. Her BMI decreased to 39.3 kg/m2 at the 1-year follow-up, with improved metabolic parameters.<br>
CONCLUSIONS: This case highlights the importance of thorough preoperative evaluation when performing LSG in patients with gastric diverticulum. Accurate endoscopic measurement of the diverticulum’s location aids in determining the optimal resection line, ensuring surgical safety and efficacy. Surgeons should remain vigilant when encountering such anatomical variations to optimize outcomes in bariatric surgery.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2045-23221512025Coronary cross-sectional area stenosis severity determined using coronary CT highly correlated with coronary functional flow reserve: a pilot study26737ENTakutoKoumotoDivision of Radiation, Okayama Heart ClinicShozoKusachiOkayama University Graduate School of Health SciencesTakumiTomiyaDivision of Cardiovascular Intervention, Okayama Heart ClinicTakuyaAkagiDivision of Cardiovascular Intervention, Okayama Heart ClinicHiroshiKawamuraDivision of Cardiovascular Medicine, Okayama Heart ClinicSatoshiHirohataOkayama University Graduate School of Health SciencesHirosukeYamajiDivision of Cardiovascular Medicine, Okayama Heart ClinicTakashiMurakamiDivision of Cardiovascular Medicine, Okayama Heart ClinicShigeshiKamikawaDivision of Cardiovascular Intervention, Okayama Heart ClinicMasaakiMurakamiDivision of Cardiovascular Intervention, Okayama Heart ClinicFractional flow reserve (FFR) is the gold standard for assessing the physiological significance of coronary stenosis. We examined the potential correlation between digitally measured coronary cross-sectional area stenosis using coronary computed tomography (CT) angiography and FFR. We analyzed data of 32 consecutive patients with stenoses who underwent invasive FFR determination. The cross-sectional area was assessed using 128-slice coronary detector-based spectral CT angiography. Power analysis revealed that the sample size enabled the detection of an area under the receiver operating characteristic (ROC) curve (AUC) of 0.90. FFR ≤ 0.8 and > 0.8 were defined as FFR-positive and FFR-negative, respectively. Intra- and interobserver differences were negligible. Percentage cross-sectional area stenosis was calculated as 100 × (A−B)/A, where A is the cross-sectional area at non-stenotic pre-stenotic segment and B is the cross-sectional area of the most severe stenotic lesion. AUC indicated that percentage cross-sectional area stenosis effectively discriminated between FFR-positive and FFR-negative cases, yielding a sensitivity of 0.882 and specificity of 0.933 at a cutoff of 50% area reduction, with an AUC of 0.976. Lesions with less than 45% cross-sectional area stenosis on coronary CT angiography were not FFR-positive. When ROC analysis was conducted for lesion characteristics, AUC did not significantly improve. In conclusion, the percent coronary cross-sectional area stenosis measured using coronary CT angiography distinguished between FFR-positive and FFR-negative lesions with high accuracy. The severity of coronary cross-sectional area stenosis determined using CT angiography is clinically useful for predicting FFR.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2666-33416S12025Basic biology is not just “for the birds”: how avian studies have informed us about vertebrate reproduction712ENGeorge E.BentleyDepartment of Integrative Biology and Helen Wills Neuroscience Institute, University of California at BerkeleySayakaAizawaGraduate School of Natural Science and Technology, Okayama UniversityAvian reproductive physiology has been studied for centuries, largely because of the importance of birds as food animals. It is likely that the ubiquity and ease of access to domesticated chickens led to them being used in some of the first experiments on transplantation of endocrine structures―in this case, the testes. Since then, study of seasonal changes in reproductive physiology (photoperiodism) in different orders of bird species has led to advances in the understanding of endocrine regulation of reproductive physiology and behavior. These include mechanisms of adult neuroplasticity, sexual selection, sperm competition, stress physiology, and circadian physiology. Here, we focus mainly on the discovery in birds of a neuropeptide named gonadotropin-inhibitory hormone that mostly has inhibitory effects on reproduction. This hormone has since been shown to exist in all mammals studied to date, including humans (it is known as RFamide-related peptide in mammals). We discuss the history and implications of avian studies on gonadotropin-inhibitory hormone/RFamide-related peptide for human reproductive biology.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2210-26121262025Rare internal hernia following pancreatoduodenectomy: A case report110673ENTeruyukiTsujiiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKoseiTakagiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYasuoNagaiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuyaYasuiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomokazuFujiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesIntroduction: Pancreatoduodenectomy (PD) is a complex procedure with a high morbidity rate. Internal hernia following PD is a rare but potentially life-threatening complication. Herein, we describe a rare case of internal hernia after PD.<br>
Presentation of case: A 76-year-old man who underwent subtotal stomach-preserving PD 7 years ago presented with vomiting and abdominal pain. Abdominal computed tomography revealed an internal hernia. Because conservative treatment failed, surgical intervention was performed. Intraoperative findings revealed efferent loop herniation in the space between the afferent loop near the Braun anastomosis and transverse mesocolon. The hernia was repositioned and the mesenteric defect was closed.<br>
Discussion: This is an extremely rare case of an internal hernia that developed 7 years after PD. As conservative management provides a little chance for improvement, precise diagnosis and prompt re-intervention are essential for the management of internal hernia. In this case, the hernial orifice developed in the space between the afferent and efferent loops and the transverse mesocolon. Internal hernia could be a differential diagnosis in patients with ileus after PD.<br>
Conclusion: This study provided a detailed description of an extremely rare case of internal hernia following PD. Therefore, internal hernias should be considered in patients undergoing PD.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama0022-510X4722025Clinical, neuroimaging and genetic findings in the Japanese case series of CLCN2-related leukoencephalopathy123486ENKentaOrimoDepartment of Neurology, Graduate School of Medicine, The University of TokyoTakashiMatsukawaDepartment of Neurology, Graduate School of Medicine, The University of TokyoAkihikoMitsutakeDepartment of Neurology, Graduate School of Medicine, The University of TokyoTakuseiChoDepartment of Neurology, Graduate School of Medicine, The University of TokyoHiroyaNaruseDepartment of Neurology, Graduate School of Medicine, The University of TokyoYoshioSakiyamaDivision of Neurology, First Department of Integrated Medicine, Saitama Medical Center, Jichi Medical UniversityKenshoSumiDepartment of Neurology, Mitsui Memorial HospitalNaohiroUchioDepartment of Neurology, Mitsui Memorial HospitalAkaneSatakeDepartment of Neurology, Fuefuki Central HospitalYoshihisaTakiyamaDepartment of Neurology, Fuefuki Central HospitalTakuyaMatsushitaDepartment of Neurology, Kochi Medical School, Kochi UniversityYosukeOmaeGenome Medical Science Project, National Center for Global Health and MedicineYosukeKawaiGenome Medical Science Project, National Center for Global Health and MedicineKatsushiTokunagaGenome Medical Science Project, National Center for Global Health and MedicineJunMitsuiDepartment of Precision Medicine Neurology, Graduate School of Medicine, The University of TokyoHiroyukiIshiuraDepartment of Neurology, Graduate School of Medicine, The University of TokyoShojiTsujiDepartment of Neurology, Graduate School of Medicine, The University of TokyoTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of TokyoBiallelic loss-of-function variants in CLCN2 lead to CLCN2-related leukoencephalopathy (CC2L), also called leukoencephalopathy with ataxia (LKPAT). CC2L is characterized clinically by a spectrum of clinical presentations including childhood- to adult-onset mild ataxia, spasticity, cognitive decline, and vision loss as well as typical MRI findings of symmetrical high signal intensities on the DWIs/T2WIs of the middle cerebellar peduncles (MCPs). We searched for pathogenic variants of CLCN2 in a case series of undiagnosed leukoencephalopathy accompanied by MCP signs, which led to the identification of four Japanese patients with CC2L. All the patients carried at least one allele of c.61dupC (p.Leu21Profs*27) in CLCN2, including compound heterozygosity with either the novel pathogenic variant c.983 + 2 T > A or the previously reported pathogenic variant c.1828C > T (p.Arg610*). Of note, all the four previously reported cases from Japan also harbored c.61dupC, and no reports of this variant have been documented from outside Japan. The allele frequency of c.61dupC in the Japanese population is 0.002152, raising the possibility of a relatively high prevalence of CC2L in Japan. Patients in this study developed symptoms after the age of 30, and demonstrated neurological signs including cerebellar ataxia, pyramidal signs, and mild cognitive impairment, consistent with previous reports. One male patient had two children, supporting preserved fertility, and another patient had calcifications in the cerebral and cerebellar surfaces. These findings provide valuable insights into the broader clinical and genetic spectra of CC2L in the Japanese population, and emphasize the importance of considering this disease in the differential diagnoses of leukoencephalopathy with MCP signs.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2950-133482025Downward hyperinflation of the native lung after right single lung transplantation for COPD: A case report highlighting diaphragmatic mobility100268ENShinTanakaDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTsuyoshiRyukoDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYasuakiTomiokaDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazuhikoShienDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenSuzawaDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKentarohMiyoshiDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMikioOkazakiDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSeiichiroSugimotoDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinichiToyookaDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesA 60-year-old male with COPD underwent right single lung transplantation. Despite progressive hyperinflation of the native left lung, the transplanted lung maintained function, as downward expansion of the left lung displaced the diaphragm without compressing the mediastinum. This suggests diaphragm mobility, aided by the absence of the liver beneath the left diaphragm, contributes to favorable outcomes in right single lung transplantation by preventing mechanical compression of the transplanted lung.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025A Case of Surgical Treatment for a Large Pulmonary Artery Aneurysm with a Quadricuspid Pulmonary Valve317320ENKeiMoriokaDepartment of Cardiovascular Surgery, Okayama University HospitalYosukeKurokoDepartment of Cardiovascular Surgery, Okayama University HospitalSachikoKadowakiDepartment of Cardiovascular Surgery, Okayama University HospitalJunkoKobayashiDepartment of Cardiovascular Surgery, Okayama University HospitalYasuhiroKotaniDepartment of Cardiovascular Surgery, Okayama University HospitalShingoKasaharaDepartment of Cardiovascular Surgery, Okayama University HospitalCase Report10.18926/AMO/69160A 65-year-old man was referred to our hospital for the annual assessment of the diameter and dilation of a pulmonary artery (PA) aneurysm. He had a small ventricular septal defect (VSD) that had closed naturally. Echocardiography revealed a dilated main PA, mild pulmonary regurgitation and no VSD. Computed tomography confirmed the dilation of the main PA (66.7×47.8 mm), right PA (37.1×32.9 mm), and left PA (36.7×34.0 mm). The patient underwent pulmonary artery replacement using a prosthetic vascular graft. A quadricuspid pulmonary valve was identified intraoperatively. Early surgical intervention could help to prevent rupture and dissection of PA aneurysms.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025Mimicking Contralateral Pneumothorax during Thoracoscopic Bullectomy Associated with Intraoperative Hyperinflation of a Large Bulla in an Obese Patient311315ENKeiMatsubaraDepartment of Thoracic Surgery, Hiroshima City Hiroshima Citizens HospitalKeiMatsubaraDepartment of Thoracic Surgery, Hiroshima City Hiroshima Citizens HospitalYutakaHiranoDepartment of Thoracic Surgery, Hiroshima City Hiroshima Citizens HospitalToshiyaFujiwaraDepartment of Thoracic Surgery, Hiroshima City Hiroshima Citizens HospitalCase Report10.18926/AMO/69159A 55-year-old obese Japanese male with left pneumothorax presented to our hospital. Bilateral pulmonary emphysema was confirmed. Persistent air leakage was observed, and a thoracoscopic bullectomy was performed. Although the thoracoscopic bullectomy was completed uneventfully, pre-extubation chest X-ray imaging indicated hyper-lucency occupying the right upper part of the thoracic cavity, suggesting right-sided pneumothorax. CT imaging indicated a right-upper-lobe expanded bulla. Extubation was performed, and the hyperinflated bulla gradually deflated. Careful management of bulla expansion and respiratory status may be necessary for patients with obesity and large bullae, especially in one-lung ventilation cases.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025A Rare Presentation of Pneumonic-Type Adenocarcinoma Hidden behind Empyema305309ENSatoruSenooDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterEitoNimanDepartment of General Thoracic Surgery, National Hospital Organization Fukuyama Medical CenterRyokoTsujiDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterKoheiTakataDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterShunsukeMatsumoriDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterFumikaMuranoDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterYukaSugisakiDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterHirokiOmoriDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterAkihikoTaniguchiDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterRikaOmoteDepartment of Diagnostic Pathology, National Hospital Organization Fukuyama Medical CenterEikiIchiharaDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterKenjiTakahashiDepartment of General Thoracic Surgery, National Hospital Organization Fukuyama Medical CenterToshiakiOkadaDepartment of Respiratory Medicine, National Hospital Organization Fukuyama Medical CenterCase Report10.18926/AMO/69158Pneumonic-type adenocarcinoma (P-ADC) can closely mimic pneumonia. We report a P-ADC initially diagnosed as pneumonia which developed into a pulmonary abscess and empyema. A 50-year-old Japanese male diagnosed with pneumonia, pulmonary abscess, and empyema was administered antibiotics and a chest tube for drainage, which improved his symptoms and blood test results. However, chest computed tomography showed an enlarged infiltrative shadow. The patient underwent bronchoscopy and was diagnosed with an adenocarcinoma. This case highlights the importance of considering P-ADC in differential diagnoses when a pneumonia-like shadow enlarges post-empyema treatment. Diagnostic and clinical tests, e.g., bronchoscopy, should be performed in such cases.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025Pulmonary Calcium Phosphate Cement Embolism After Percutaneous Vertebroplasty for Thoracic Vertebrae Fractures299303ENRuibinFengDepartment of Orthopedics, the Ninth Affiliated Hospital of Guangxi Medical UniversityBikangZhuDepartment of Orthopedics, the Ninth Affiliated Hospital of Guangxi Medical UniversityDanyunWeiDepartment of Orthopedics, the Ninth Affiliated Hospital of Guangxi Medical UniversityDingjiaoZhuDepartment of Radiology, the Ninth Affiliated Hospital of Guangxi Medical UniversityCairuChenDepartment of Orthopedics, the Ninth Affiliated Hospital of Guangxi Medical UniversityCase Report10.18926/AMO/69157Pulmonary cement embolism (PCE) is a rare but severe complication following percutaneous vertebroplasty (PVP). Calcium phosphate cement (CPC) has emerged as an alternative to traditional materials for vertebral augmentation. There appear to be no established guidelines for managing symptomatic PCE, and there is scarce literature on CPC embolisms. This is a first report of a case of pulmonary CPC embolism following PVP. The patient, a 63-year-old Chinese female, was administered anticoagulant treatment and achieved a satisfactory outcome. Her case highlights the severe potential morbidity associated with CPC leakage and emphasizes the efficacy of anticoagulant treatment for managing pulmonary CPC embolisms.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025Effectiveness of Pallidal Stimulation for Dystonic Storm and Subsequent Ssevere Posterior Reversible Encephalopathy Syndrome in a Patient with GNAO1 Variant293297ENKojiKawaiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTatsuyaSasakiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShunTanimotoDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomoyaSaijoDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSusumuSasadaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomoyukiAkiyamaDepartment of Pediatric Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakuyaHiraideDepartment of Biochemistry, Hamamatsu University School of MedicineHirotomoSaitsuDepartment of Biochemistry, Hamamatsu University School of MedicineShotaTanakaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/69156GNAO1 variant affects primarily the brain and neurodevelopment, leading to a range of motor disorders including seizures beginning in infancy and involuntary movements such as dyskinesia and dystonia. Our patient, a 15-year-old Japanese female, began exhibiting involuntary movements at age 4. A de novo missense mutation (NM_020988.3: c.228C>G, NP_066268.1: p.(Asn76Lys)) in the GNAO1 gene was identified when the patient was 15, and during the same year she developed influenza pneumonia, accompanied by dystonic storm. She required intensive care with mechanical ventilation and underwent a tracheostomy. She also developed posterior reversible encephalopathy syndrome. Globus pallidal stimulation was administered, leading to an improvement in the dystonic storm. Early consideration of globus pallidal stimulation is recommended when treating difficult-to-manage dystonic storms.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025Parieto-Occipital Disconnection for Drug-Resistant Parieto-Occipital Lobe Epilepsy: A Case Report and Surgical Technique287292ENShunTanimotoDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTatsuyaSasakiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKojiKawaiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomoyaSaijoDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKyoheiKinDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSusumuSasadaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShotaTanakaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/69155We report a case of drug-resistant parieto-occipital lobe epilepsy successfully treated with parieto-occipital disconnection (POD). An 18-year-old left-handed female, who had undergone surgery for an acute subdural hematoma at 10 months of age, developed drug-resistant epilepsy at age 15. Despite antiepileptic drug treatment, her seizures remained uncontrolled, and at age 18 she was referred to our hospital for evaluation. Magnetic resonance imaging (MRI) revealed atrophy in the left occipital and parietal lobes. Ictal electroencephalography (EEG) confirmed occipital onset of seizures without temporal lobe involvement. She had pre-existing homonymous hemianopsia. POD surgery was performed, carefully preserving the temporal lobe structures. Postoperatively, she experienced transient right-sided paresis, which fully resolved, and achieved complete seizure control at 3 years without memory loss. This case demonstrates that POD, a rare surgical approach, is a viable option for parieto-occipital lobe epilepsy, effectively controlling seizures while minimizing functional impairment in the absence of temporal lobe involvement.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025Anterior Uveitis Secondary to an Infected Postoperative Maxillary Cyst283286ENYutaImamuraDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYusukeShiodeDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShuheiKimuraDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMioHosokawaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyoMatobaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiKanzakiDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHiroyaKindoDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTetsuroMoritaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAyaMuraiDepartment of Otolaryngology-Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMizuoAndoDepartment of Otolaryngology-Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiMorizaneDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/69154A 76-year-old man presented with right eyelid swelling and deteriorated vision. Examination revealed anterior uveitis with hypopyon and a visual acuity of 20/2,000 in the right eye, with no abnormalities in the left. Computed tomography revealed enlargement of the right maxillary sinus and internal fluid accumulation, suggesting a postoperative maxillary cyst (POMC). Nasal endoscopic surgery drained the pus by opening the lower wall of the maxillary cyst. Following the procedure, intraocular inflammation resolved, and visual acuity in the right eye improved to 24/20. This is the first reported case of uveitis secondary to POMC.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025Long-Term Survival Following Extended Cholecystectomy for Synchronous Gallbladder and Regional Lymph Node Metastasis of Lung Adenocarcinoma, with Subsequent Pulmonary Lobectomy279282ENMaoYoshikawaDepartment of General Thoracic Surgery, Okayama University HospitalHiroyukiTaoDepartment of Thoracic Surgery, Japanese Red Cross Society Himeji HospitalCase Report10.18926/AMO/69153An 80-year-old male underwent an extended cholecystectomy for node-positive gallbladder adenocarcinoma. Two weeks later, hemoptysis revealed a left hilar tumor obstructing the bronchus, which was diagnosed as adenocarcinoma. Three months post-cholecystectomy, a left upper pulmonary lobectomy was performed. Histological similarity and positive thyroid transcription factor-1 (TTF-1) immunostaining in both tumors confirmed lung adenocarcinoma with gallbladder metastasis. Despite the generally poor prognosis for gallbladder metastasis from lung cancer, the patient achieved 3 years of survival. Patients with isolated synchronous gallbladder metastasis from lung cancer may benefit from oligometastasectomy.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025Femoral and Global Femoral Offset, but not Anteroposterior Offset, to Improve Postoperative Outcomes Following Total Hip Arthroplasty: Considerations Independent of the Contralateral Side269278ENNorioImaiDivision of Comprehensive Musculoskeletal Medicine, Niigata University Graduate School of Medical and Dental SciencesYukiHiranoDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesDaisukeHommaDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesYukiEndoDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesYojiHorigomeDivision of Comprehensive Musculoskeletal Medicine, Niigata University Graduate School of Medical and Dental SciencesHayatoSuzukiDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesHiroyukiKawashimaDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesOriginal Article10.18926/AMO/69152The global femoral offset (the sum of the acetabular and femoral offsets) influences outcomes after total hip arthroplasty (THA). The optimal offset using plain radiographs has been reported, but internal and external rotations of the hip affect the offset value, producing unclear results when the nonsurgical side is not intact. We investigated the relationship between a functional hip score, i.e., the Harris Hip Score (HHS) and its effect on the post-THA anteroposterior and lateral offsets, and we sought to identify the optimal offset value. The cases of 158 patients with hemilateral hip osteoarthritis who underwent THA at a single center were retrospectively analyzed in this cross-sectional study. Three-dimensional pelvic and femoral models generated from computed tomography were used to examine several parameters, and the results revealed a significant binomial correlation among the modified HHS and femoral and global femoral offsets, with maximum values of 21.3 mm and 40 mm/100 cm body height, respectively. Pelvic and femoral parameters were measured and evaluated via alignment with a specific coordinate system. Our findings indicate that preoperative planning using these parameters may improve postoperative hip function, even when the nonoperative side is unsuitable for use as a reference, as in bilateral hip osteoarthritis cases.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7942025Organ Donation after Extracorporeal Cardiopulmonary Resuscitation: Clinical and Ethical Perspectives221229ENTetsuyaYumotoDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityHiromichiNaitoDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTakashiHongoDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTakafumiObaraDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYoshinoriKosakiDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKoheiAgetaDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTsuyoshiNojimaDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKoheiTsukaharaDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityAtsunoriNakaoDepartment of Emergency, Critical Care and Disaster Medicine, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityReview10.18926/AMO/69147Extracorporeal cardiopulmonary resuscitation (ECPR) has evolved into a life-saving therapy for select cardiac arrest patients, yet a growing body of evidence suggests it also holds promise as a bridge to organ donation in non-survivors. This review explores the clinical outcomes, ethical complexities, and evolving policies surrounding organ donation after ECPR. We summarize recent international and Japanese data demonstrating favorable graft function from ECPR donors, with the exception of lung transplantation. The ethical challenges ― particularly those involving brain death determination on extracorporeal membrane oxygenation and adherence to the dead donor rule ― are discussed in the context of Japan’s recent regulatory reforms. Additionally, we highlight the importance of structured end-of-life communication through multidisciplinary team meetings in facilitating ethically sound transitions from rescue efforts to donation pathways. Moving forward, improvements in donor management, standardized legal frameworks, and public and professional education are essential to optimizing the life-saving and life-giving potential of ECPR.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama1351-51013212025Lyme neuroborreliosis in Japan: Borrelia burgdorferi sensu lato as a cause of meningitis of previously undetermined etiology in hospitalized patients outside of the island of Hokkaido, 2010–2021e70005ENMasayukiOhiraDepartment of Clinical Laboratory and Internal Medicine, National Center of Neurology and PsychiatryAiTakanoDepartment of Veterinary Medicine, Joint Faculty of Veterinary Medicine, Yamaguchi UniversityKentaroYoshiNational Research Center for the Control and Prevention of Infectious Diseases, Nagasaki UniversityAkiraAraiDepartment of Neurology, Aomori Prefectural Central HospitalYashuhiroAsoDepartment of Neurology, Oita Prefectural HospitalRikiyaFurutaniDepartment of Neurology, National Hospital Organization, Shinshu Ueda General HospitalTadanoriHamanoDepartment of Neurology, University of Fukui HospitalIkukoTakahashi‐IwataDepartment of Neurology, Hokkaido University HospitalChikakoKanekoDepartment of Neurology, Southern Tohoku General HospitalTohruMatsuuraDivision of Neurology, Jichi Medical UniversityNorihisaMaedaDepartment of Neurology, National Hospital Organization Beppu Medical CenterHidetoNakajimaDepartment of Neurology, Nihon University Itabashi HospitalKatsuroShindoDepartment of Neurology, Kurashiki Central HospitalToshihikoSuenagaDepartment of Neurology, Tenri HospitalKazumaSugieDepartment of Neurology, Nara Medical University HospitalYasuhiroSuzukiDepartment of Neurology, National Hospital Organization Asahikawa Medical CenterToruYamashitaDepartment of Neurology, Okayama University HospitalFrederick J.AnguloVaccines and Antivirals Medical Affairs, Pfizer VaccinesJuanitaEdwardsVaccines and Antivirals Medical Affairs, Pfizer VaccinesCody MatthewBenderVaccines and Antivirals Medical Affairs, Pfizer VaccinesLisa R.HarperVaccines and Antivirals Medical Affairs, Pfizer VaccinesYoshikazuNakayamaVaccines Medical Affairs, Pfizer Japan IncShuheiItoVaccines Medical Affairs, Pfizer Japan IncAndreasPilzVaccines and Antivirals Medical Affairs, Pfizer VaccinesJames H.StarkVaccines and Antivirals Medical Affairs, Pfizer VaccinesJennifer C.MoïsiVaccines and Antivirals Medical Affairs, Pfizer VaccinesHidehiroMizusawaDepartment of Neurology, National Center of Neurology and PsychiatryMasakiTakaoDepartment of Clinical Laboratory and Internal Medicine, National Center of Neurology and PsychiatryBackground and Purpose: Clinical manifestations of Lyme borreliosis (LB), caused by Borrelia burgdorferi sensu lato (Bbsl), include erythema migrans, Lyme neuroborreliosis (LNB), carditis, and arthritis. LB is a notifiable disease in Japan with <30 surveillance-reported LB cases annually, predominately from Hokkaido Prefecture. However, LB, including LNB, may be under-diagnosed in Japan since diagnostic tests are not readily available. We sought to determine if LNB could be a cause of previously undiagnosed encephalitis or meningitis in Japan.<br>
Methods: Investigators at 15 hospitals in 10 prefectures throughout Japan retrieved serum and/or cerebrospinal fluid (CSF) samples collected in 2010–2021 from 517 patients hospitalized with encephalitis or meningitis which had an etiology that had not been determined. Samples were tested for Bbsl-specific antibodies using ELISA and Western blot tests. In alignment with the European Union LNB case definition, a confirmed LNB case had CSF pleocytosis and intrathecal production of Bbsl-specific antibodies and a probable LNB case had a CSF sample with pleocytosis and Bbsl-specific antibodies.<br>
Results: LNB was identified in three hospitalized patients with meningitis of previously undetermined etiology: a male resident of Aomori Prefecture was a confirmed LNB case, and two female residents of Oita Prefecture were probable LNB cases. None of the patients with confirmed or probable LNB had traveled in the month prior to symptom onset and none had samples previously tested for LB.<br>
Conclusion: The identification of previously undiagnosed LNB cases indicates a need for enhanced disease awareness in Japan, particularly beyond Hokkaido Island, and more readily available LB diagnostic testing.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-09041372025Tongue Schwannoma at the Median Inferior Surface in the Elderly: A Case Reporte70506ENKihoFukushimaDepartment of Oral and Maxillofacial Surgery, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKishoOnoDepartment of Oral and Maxillofacial Surgery, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKyoichiObataDepartment of Oral and Maxillofacial Surgery, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityIzumiYamamotoDepartment of Oral and Maxillofacial Surgery, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYukiKunisadaDepartment of Oral and Maxillofacial Surgery, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityHirokazuYutoriDepartment of Oral and Maxillofacial Surgery, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversitySoichiroIbaragiDepartment of Oral and Maxillofacial Surgery, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityWe report the extremely rare case of an atypical schwannoma that occurred at the median inferior surface of the tongue in an elderly patient. We performed an excisional biopsy to achieve a definitive diagnosis. Based on the histopathological findings, we diagnosed a schwannoma (mixed type, Antoni A/B).No potential conflict of interest relevant to this article was reported.Japanese Society of Internal MedicineActa Medica Okayama0918-291864122025Subacute Upper Motor Neuron Dysfunction Possibly Associated with the Anti-GM1 Autoantibody19001905ENSoOkuboDepartment of Neurology, Graduate School of Medicine, The University of TokyoMeikoMaedaDepartment of Neurology, Graduate School of Medicine, The University of TokyoKazutoKatsuseDepartment of Neurology, Graduate School of Medicine, The University of TokyoHiroyukiIshiuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYuichiroShirotaDepartment of Neurology, Graduate School of Medicine, The University of TokyoMasashiHamadaDepartment of Neurology, Graduate School of Medicine, The University of TokyoWataruSatakeDepartment of Neurology, Graduate School of Medicine, The University of TokyoTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of TokyoAnti-GM1 antibodies are associated with Guillain-Barré syndrome (GBS), primarily peripheral neuropathy. However, there are cases of anti-GM1 IgG antibody-positive GBS with upper motor neuron (UMN) signs. We herein report a case of gastrointestinal infection followed by subacute gait disturbance with predominant signs of UMN on a neurological examination. The serum and cerebrospinal fluid tests were positive for anti-GM1 and anti-asialo-GM1 IgG antibodies. An electrophysiological evaluation revealed normal nerve conduction and prolonged central motor conduction times. No magnetic resonance imaging abnormalities were observed. The symptoms improved with treatment, which was accompanied by decreased antibody titers. This case highlights the fact that anti-GM1 IgG-associated disorders may present with predominant UMN involvement.No potential conflict of interest relevant to this article was reported.Japan Surgical SocietyActa Medica Okayama2198-77931112025A Case of Omental Bleeding as a Result of Segmental Arterial Mediolysis Treated Successfully by Laparoscopic Partial Omentectomycr.25-0262ENYudaiMimataDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesNobuhikoKanayaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshitakaKondoDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHitoshiMinagiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshihikoKakiuchiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinjiKurodaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKunitoshiShigeyasuDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShunsukeKagawaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesINTRODUCTION: Segmental arterial mediolysis (SAM) is a rare, non-atherosclerotic, non-inflammatory arteriopathy characterized by lysis of the arterial media, leading to aneurysm formation and possible rupture. Although visceral arteries are typically involved, SAM-induced omental bleeding is extremely uncommon. While transcatheter arterial embolization (TAE) has been reported, surgical resection offers both definitive hemostasis and histopathological confirmation.<br>
CASE PRESENTATION: A 56-year-old man presented with upper abdominal pain without a history of trauma. Contrast-enhanced CT revealed a hematoma and fusiform dilation of an omental artery, suggesting omental hemorrhage. As he was hemodynamically stable, initial conservative management was chosen. However, a follow-up CT on day 7 demonstrated aneurysm enlargement, prompting laparoscopic partial omentectomy. Intraoperative findings included a 5-cm hematoma in the central omentum. Histopathological examination showed vacuolization of the tunica media and loss of the internal elastic lamina, confirming the diagnosis of SAM. The patient had an uneventful postoperative course and was discharged on the 3rd postoperative day.<br>
CONCLUSIONS: This rare case of SAM-related omental bleeding was successfully treated with laparoscopic partial omentectomy. Tailored treatment strategies including laparoscopic surgery are essential for optimal outcomes in SAM.<br>No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1434-516169122024Association study of GBA1 variants with MSA based on comprehensive sequence analysis -Pitfalls in short-read sequence analysis depending on the human reference genome-613621ENKentaOrimoDepartment of Neurology, Graduate School of Medicine, The University of TokyoJunMitsuiDepartment of Precision Medicine Neurology, Graduate School of Medicine, The University of TokyoTakashiMatsukawaDepartment of Neurology, Graduate School of Medicine, The University of TokyoMasakiTanakaInstitute of Medical Genomics, International University of Health and WelfareJunkoNomotoInstitute of Medical Genomics, International University of Health and WelfareHiroyukiIshiuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYosukeOmaeGenome Medical Science Project, National Center for Global Health and MedicineYosukeKawaiGenome Medical Science Project, National Center for Global Health and MedicineKatsushiTokunagaGenome Medical Science Project, National Center for Global Health and MedicineNCBN Controls WGS ConsortiumTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of TokyoShojiTsujiDepartment of Neurology, Graduate School of Medicine, The University of TokyoMultiple system atrophy (MSA) is a neurodegenerative disorder characterized by various combinations of autonomic failure, parkinsonism, and cerebellar ataxia. To elucidate variants associated with MSA, we have been conducting short-read-based whole-genome sequence analysis. In the process of the association studies, we initially focused on GBA1, a previously proposed susceptibility gene for MSA, to evaluate whether GBA1 variants can be efficiently identified despite its extraordinarily high homology with its pseudogene, GBA1LP. To accomplish this, we conducted a short-read whole-genome sequence analysis with alignment to GRCh38 as well as Sanger sequence analysis and compared the results. We identified five variants with inconsistencies between the two pipelines, of which three variants (p.L483P, p.A495P–p.V499V, p.L483_M489delinsW) were the results of misalignment due to minor alleles in GBA1P1 registered in GRCh38. The miscalling events in these variants were resolved by alignment to GRCh37 as the reference genome, where the major alleles are registered. In addition, a structural variant was not properly identified either by short-read or by Sanger sequence analyses. Having accomplished correct variant calling, we identified three variants pathogenic for Gaucher disease (p.S310G, p.L483P, and p.L483_M489delinsW). Of these variants, the allele frequency of p.L483P (0.003) in the MSA cases was higher than that (0.0011) in controls. The meta-analysis incorporating a previous report demonstrated a significant association of p.L483P with MSA with an odds ratio of 2.85 (95% CI; 1.05 – 7.76, p = 0.0400).No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama1201-97121562025Recurrent diffuse panbronchiolitis after lung transplantation: Off-label use of inhaled tobramycin for Pseudomonas aeruginosa control in a transplant recipient107913ENShinTanakaDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTuyoshiRyukoDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYasuakiTomiokaDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazuhikoShienDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenSuzawaDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKentarohMiyoshiDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalMikioOkazakiDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSeiichiroSugimotoDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinichiToyookaDepartment of General Thoracic and Breast and Endocrinological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesObjectives: This report highlights a clinical case of recurrent diffuse panbronchiolitis (DPB) after bilateral lung transplantation (LTx), with a focus on the therapeutic impact of off-label inhaled tobramycin solution for inhalation (TSI) in managing Pseudomonas aeruginosa colonization.<br>
Methods: A Japanese woman with a history of DPB experienced disease recurrence following bilateral LTx. Persistent colonization by P. aeruginosa and recurrent respiratory symptoms were observed. Off-label TSI therapy, commonly used in cystic fibrosis, was introduced. Clinical response was assessed through radiologic imaging, bronchoscopy, and microbiological cultures.<br>
Results: TSI administration led to significant clinical and radiological improvement. P. aeruginosa was eradicated from sputum cultures within one month and remained absent throughout six months of follow-up. No hospitalizations or adverse events were reported during therapy.<br>
Conclusion: This case suggests the potential of TSI as a therapeutic approach for managing recurrent DPB and indicates its role in stabilizing post-transplant outcomes. Further studies may clarify its efficacy and expand its application in broader DPB management strategies.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2363-90241112025Non-convulsive status epilepticus as a cause of delayed emergence after a thoracic surgery: a case report30ENYusukeIritaniDepartment of Anesthesiology, Okayama Red Cross HospitalMakikoTaniDepartment of Anesthesiology and Resuscitology, Graduate School of Medicine Dentistry and Pharmaceutical Sciences, Okayama UniversityShinjiIgaDepartment of Anesthesiology and Resuscitology, Okayama University HospitalHiroshiMorimatsuDepartment of Anesthesiology and Resuscitology, Graduate School of Medicine Dentistry and Pharmaceutical Sciences, Okayama UniversityNon-convulsive status epilepticus (NCSE) is an electrical discharge which occurs without prominent motor symptoms. NCSE is one of the causes of delayed emergence from anesthesia; however, as far as we know, previous reports of postoperative NCSE were related to patients after neurological surgery. Herein, we report a case of an elderly male who developed initial NCSE after thoracic surgery. The patient remained unresponsive and developed hemiplegia after lung resection, and then the symptoms fluctuated between better and worse. Metabolic disorders and stroke were ruled out, and NCSE was diagnosed by magnetic resonance imaging (MRI) and electroencephalography (EEG). NCSE occurred in a patient who had no predisposing factors or underwent non-neurological surgery. When anesthesiologists encounter delayed emergence, NCSE should be listed as a differential diagnosis and examined by MRI and EEG.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2692-4609612025A Case of Gastric Atypical Lipomatous Tumor/Well‐Differentiated Liposarcoma With Endoscopic Morphological Changese70146ENRikaOmoteDepartment of Diagnostic Pathology, NHO Fukuyama Medical CenterShizumaOmoteDepartment of Internal Medicine, Fukuyama Minami HospitalHiroshiSonobeDepartment of Diagnostic Pathology, NHO Fukuyama Medical CenterRyosukeHamanoDepartment of Surgery, NHO Fukuyama Medical CenterTatsuyaToyokawaDepartment of Gastroenterology, NHO Fukuyama Medical CenterShinyaOtsukaDepartment of Surgery, NHO Fukuyama Medical CenterTakehiroTanakaDepartment of Pathology and Oncology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHiroyukiYanaiDepartment of Pathology, Okayama University HospitalMasaruInagakiDepartment of Surgery, NHO Fukuyama Medical CenterHidetakaYamamotoDepartment of Pathology and Oncology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAtypical lipomatous tumor/well-differentiated liposarcoma is a locally aggressive mesenchymal neoplasm composed of adipocytes and stromal cells. Gastric cases are exceedingly rare, and their malignant potential remains unclear. We report a case of a woman in her 60s who was found to have multiple submucosal tumor-like lesions of the stomach. Over time, the tumors increased in size, requiring a laparoscopic partial gastrectomy. Histological examination revealed a tumor composed of both fatty tissue and fibrous stroma with nuclear atypia. Immunohistochemistry showed positivity for CDK4 and MDM2, and fluorescence in situ hybridization confirmed MDM2 amplification, leading to a diagnosis of atypical lipomatous tumor/well-differentiated liposarcoma. This case presented an unusual gastric manifestation, with multiple submucosal tumor-like lesions on endoscopy and exhibiting progressive morphological changes over several years.No potential conflict of interest relevant to this article was reported.MDPI AGActa Medica Okayama2077-038314102025Endoscopic Bridging Stent Placement Improves Bile Leaks After Hepatic Surgery3381ENTaisukeObataDepartment of Gastroenterology and Hepatology, Okayama University HospitalKazuyukiMatsumotoDepartment of Gastroenterology and Hepatology, Okayama University HospitalKeiHaradaDepartment of Gastroenterology and Hepatology, Okayama University HospitalNaoHattoriDepartment of Gastroenterology and Hepatology, Okayama University HospitalRyosukeSatoDepartment of Gastroenterology and Hepatology, Okayama University HospitalAkihiroMatsumiDepartment of Gastroenterology and Hepatology, Okayama University Hospital, 2-5-1 Shikata-cho, Okayama 700-8558, JapanKazuyaMiyamotoDepartment of Gastroenterology and Hepatology, Okayama University HospitalHiroyukiTerasawaDepartment of Gastroenterology and Hepatology, Okayama University HospitalYukiFujiiDepartment of Gastroenterology and Hepatology, Okayama University HospitalDaisukeUchidaDepartment of Gastroenterology and Hepatology, Okayama University HospitalShigeruHoriguchiDepartment of Gastroenterology and Hepatology, Okayama University HospitalKoichiroTsutsumiDepartment of Gastroenterology and Hepatology, Okayama University HospitalMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University HospitalBackground: Endoscopic treatment is one of the first-line treatments for bile leaks after hepatic surgery. However, detailed reports of endoscopic treatment for bile leaks after hepatic resection (HR) or liver transplantation (LT) are scarce. The outcomes of endoscopic treatment for bile leaks after hepatic surgery were examined, and factors related to successful treatment were identified. Methods: A total of 122 patients underwent endoscopic treatment for bile leaks after hepatic surgery. The diagnosis of a bile leak is based on the ISGLS criteria. The decision to perform endoscopic retrograde cholangiography (ERC) is made based on the amount of drainage output, laboratory data, clinical symptoms, and CT scan findings. In our study, the site of the bile leak was assessed using ERC. Endoscopic stents were placed to bridge across the bile leak site as much as possible. Otherwise, stents were placed near the leak site. Endoscopic stents were replaced every 2–3 months until an improvement in the bile leak was observed with or without biliary strictures. The outcomes of endoscopic treatment and the factors related to clinical success were evaluated. Results: Seventy-four patients with HR and forty-eight patients with LT were treated endoscopically. Technical and clinical success was achieved in 89% (109/122) and 82% (100/122) of patients, respectively. Three (2%) patients died from uncontrollable bile leaks. Bridging stent placement (p < 0.001), coexistent percutaneous drainage (p = 0.0025), and leak severity (p = 0.015) were identified as independent factors related to the clinical success of endoscopic treatment. During a median observation period of 1162 days after the achievement of clinical success, bile leak recurrence was observed in only three cases (3%). Conclusions: Endoscopic treatment is safe and effective for bile leaks after hepatic surgery. Bridging stent placement across the leak site is the most crucial factor for clinical success.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1752-19471912025Exercise hemodynamic evaluation in the management of dasatinib-related pulmonary arterial hypertension: a case report209ENShuheiYamashitaDepartment of Cardiology, Keio University School of MedicineTakahiroHiraideDepartment of Cardiology, Keio University School of MedicineYasuyukiShiraishiDepartment of Cardiology, Keio University School of MedicineYoshinoriKatsumataDepartment of Cardiology, Keio University School of MedicineMasaharuKataokaDepartment of Cardiology, Keio University School of MedicineShogoFukuiDepartment of Rehabilitation, Keio University HospitalMichiyukiKawakamiDepartment of Rehabilitation, Keio University School of MedicineShinsukeYuasaDepartment of Cardiovascular Medicine Academic Field, Dentistry and Pharmaceutical Sciences, Okayama UniversityShinichiroOkamotoDepartment of Hematology, Keio University School of MedicineKeiichiFukudaDepartment of Cardiology, Keio University School of MedicineMasakiIedaDepartment of Cardiology, Keio University School of MedicineBackground Dasatinib-related pulmonary arterial hypertension is a rare complication of chronic therapy for hematological malignancies. Pulmonary hypertension often persists despite drug discontinuation and might require vasodilators. Normalizing pulmonary hemodynamics and avoiding the long-term use of vasodilators is challenging.<br>
Case presentation Patient was a 55-year-old Japanese man complaining of progressive dyspnea on effort and fatigue. He had a history of hypertension and chronic myeloid leukemia treated with dasatinib. He was diagnosed with dasatinib-related pulmonary arterial hypertension by a right heart catheterization at rest, demonstrating a mean pulmonary artery pressure of 31 mmHg and a normal pulmonary arterial wedge pressure of 6 mmHg. Symptoms and hemodynamics significantly improved after the discontinuation of dasatinib and the initiation of upfront combination therapy of vasodilators. An exercise right heart catheterization, performed more than 2 years after the initiation of vasodilators, showed a mean pulmonary artery pressure of 15 mmHg at rest and 29 mmHg at peak exercise (normal reference value, < 30 mmHg), suggesting normal pulmonary microcirculation. On the basis of these findings, pulmonary vasodilators were discontinued. Notably, a repeat exercise right heart catheterization demonstrated preserved pulmonary microcirculation, and the patient has remained asymptomatic for more than 2 years after discontinuing pulmonary-arterial-hypertension-targeted therapy.<br>
Conclusions The evaluation of pulmonary microcirculation by exercise right heart catheterization can be useful for withdrawing pulmonary vasodilators safely in the management of patients with dasatinib-related pulmonary arterial hypertension.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841772025Six-Year Remission With No Relapse After Four-Time Weekly Rituximab Only for Bilateral Ocular Adnexal Follicular Lymphomae88945ENToshihikoMatsuoOphthalmology, Graduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama University, and Okayama University HospitalTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityNobuharuFujiiDivision of Transfusion and Cell Therapy, Department of Hematology and Oncology, Okayama University HospitalFollicular lymphoma mostly takes an indolent course, and thus, observation with watchful waiting is a main therapeutic strategy. Recent long-term studies suggest earlier treatment with rituximab monotherapy may benefit patients by delaying the need for treatment in the later phase of exacerbation. In this study, we reported a patient with bilateral orbital follicular lymphoma who received four-time weekly rituximab monotherapy as an induction therapy only and maintained the remission for 5 years with no treatment. The patient was a 51-year-old woman who developed a right upper orbital mass and was diagnosed with follicular lymphoma grade 1 by the excisional biopsy. Two years later, at the age of 53 years, she developed a left lacrimal gland mass and underwent excision. The pathological diagnosis was follicular lymphoma grade 1. She did not have any other systemic lesions by fluorodeoxyglucose positron emission tomography. At the age of 54 years, she developed a new mass on the nasal side of the right orbit and underwent weekly rituximab monotherapy (375 mg/m2) four times a month, leading to the reduction of the mass in 3 months. Two high uptake sites on the temporal and nasal side of the right superior orbit by fluorodeoxyglucose positron emission tomography disappeared one year later at the age of 55 years. She was followed with no treatment for 6 years until the age of 60 years at the latest visit. In case of a local orbital relapse, local radiotherapy would be the standard, but rituximab monotherapy as an induction therapy only was chosen in the present patient. Rituximab monotherapy in place of local radiotherapy would be a treatment option for orbital follicular lymphoma.No potential conflict of interest relevant to this article was reported.MDPIActa Medica Okayama1422-006726142025Inhibitory Effects of Vandetanib on Catecholamine Synthesis in Rat Pheochromocytoma PC12 Cells6927ENYoshihikoItohDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKenichiInagakiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTomohiroTerasakaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityEisakuMorimotoDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTakahiroIshiiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKimitomoYamaokaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversitySatoshiFujisawaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityGain-of-function gene alterations in rearranged during transfection (RET), a receptor tyrosine kinase, are observed in both sporadic and hereditary medullary thyroid cancers (MTCs) and pheochromocytomas and paragangliomas (PPGLs). Several tyrosine kinase inhibitors (TKIs) that target RET have been proven to be effective on MTCs and PCCs. Recently, TKIs, namely, sunitinib and selpercatinib, which were clinically used to target PPGLs, have been reported to decrease catecholamine levels without reducing tumor size. Our clinical case of metastatic medullary thyroid cancer, which is associated with RET mutations undergoing treatment with vandetanib, also suggests that vandetanib can decrease catecholamine levels. Therefore, we investigated the effect of vandetanib, a representative multi-targeted TKI for RET-related MTC, on cell proliferation and catecholamine synthesis in rat pheochromocytoma PC12 cells. Vandetanib reduced viable cells in a concentration-dependent manner. The dopamine and noradrenaline levels of the cell lysate were reduced in a concentration-dependent manner. They also decreased more prominently at lower concentrations of vandetanib compared to the inhibition of cell proliferation. The RNA knockdown study of Ret revealed that this inhibitory effect on catecholamine synthesis is mainly mediated by the suppression of RET signaling. Next, we focused on two signaling pathways downstream of RET, namely, ERK and AKT signaling. Treatment with vandetanib reduced both ERK and AKT phosphorylation in PC12 cells. Moreover, both an MEK inhibitor U0126 and a PI3K/AKT inhibitor LY294002 suppressed catecholamine synthesis without decreasing viable cells. This study in rat pheochromocytoma PC12 cells reveals the direct inhibitory effects of vandetanib on catecholamine synthesis via the suppression of RET-ERK and RET-AKT signaling.No potential conflict of interest relevant to this article was reported.Japan Neurosurgical SocietyActa Medica Okayama2188-4226122025Safety of Adenosine-assisted Clipping Surgery for Unruptured Cerebral Aneurysms: Interim Results of a Single-center, Single-arm Study115119ENTomohitoHISHIKAWADepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSatoshiMURAIDepartment of Neurosurgery, Kawasaki Medical SchoolMasafumiHIRAMATSUDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunHARUMADepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiEBISUDANIDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakaoYASUHARADepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenjiSUGIUDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazuyoshiSHIMIZUDepartment of Anesthesiology and Resuscitology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKojiNAKAGAWADepartment of Cardiovascular Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAyaKIMURA-ONOCenter for Innovative Clinical Medicine, Okayama University HospitalKatsuyukiHOTTACenter for Innovative Clinical Medicine, Okayama University HospitalHiroshiMORIMATSUDepartment of Anesthesiology and Resuscitology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesIsaoDATEDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesThe aim of this single-center, single-arm study was to evaluate the safety of adenosine-assisted clipping surgery for unruptured cerebral aneurysms. Five patients underwent aneurysmal clipping during adenosine-induced hypotension at ≤60 mmHg. The mean age of patients was 63.4±8.5 years, and the mean aneurysm size was 5.3±1.1 mm. The prevalence of patients with modified Rankin Scale scores of zero 30 days after surgery was 100%. The degree of aneurysm obliteration was complete in 4 patients and residual dome in 1 patient. The mean total dosage of adenosine was 37.4±18.8 mg. The mean duration of systolic blood pressure at ≤60 mmHg was 64.2±28.3 secs. No patients exhibited paroxysmal atrial fibrillation within 24 hours after adenosine administration or elevation of high-sensitivity cardiac troponin T on postoperative day 1. There was no reduction in either motor-evoked or somatosensory-evoked potential amplitude during surgery. Adenosine-induced hypotension is a safe procedure in clipping surgery for unruptured cerebral aneurysms.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2045-23221512025Trends in the incidence of severe fever with thrombocytopenia syndrome in Japan: an observational study from 2013 to 202220715ENShinnosukeFukushimaDepartment of General Medicine, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineHidemasaAkazawaDepartment of General Medicine, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineToshihiroKoyamaDepartment of Health Data Science, Dentistry, and Pharmaceutical Sciences, Okayama University Graduate School of MedicineHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalWe aimed to determine the 10-year trend in the incidence of Severe fever with thrombocytopenia syndrome (SFTS) in Japan. This retrospective observational study used a publicly available national database. Trends in the incidence of SFTS with annual percent changes (APC) were examined using Joinpoint regression analysis with stratification by patient age, season, and region. The association between disease incidence and environmental factors was investigated using Spearman’s rank correlation. Between 2013 and 2022, there were 803 notified cases (397 males and 406 females) of SFTS, with 79.5% aged ≥ 65 years. The annual incidence rate increased continuously with an APC of 9.6%. The incidence peaked between May and June, with 80.8% of cases observed between May and October. The incidence was predominantly higher in western Japan, and the mean annual incidence rate was the highest in Miyazaki prefecture, with 0.89 per 100,000 people. Correlations between the SFTS incidence rates and environmental factors were observed in western Japan, with forest area (correlation coefficient, 0.80), followed by agricultural population rate (0.70). SFTS incidence is continuously increasing in Japan, especially among the elderly population. Environmental factors such as broader forest areas and increased agricultural population were possibly associated with the incidence.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1471-24662512025Co-occurrence of interstitial lung disease and pulmonary embolism as adverse events of adjuvant osimertinib treatment for EGFR mutant non-small cell lung cancer: a case report311ENKentaManabeDepartment of Thoracic Surgery, Okayama University HospitalKazuhikoShienDepartment of Thoracic Surgery, Okayama University HospitalShinichiFurukawaDepartment of Thoracic Surgery, Okayama University HospitalTomoyaSenoDepartment of Thoracic Surgery, Okayama University HospitalKouseiIshimuraDepartment of Thoracic Surgery, Okayama University HospitalShinTanakaDepartment of Thoracic Surgery, Okayama University HospitalKenSuzawaDepartment of Thoracic Surgery, Okayama University HospitalMikioOkazakiDepartment of Thoracic Surgery, Okayama University HospitalSeiichiroSugimotoDepartment of Thoracic Surgery, Okayama University HospitalShinichiToyookaDepartment of Thoracic Surgery, Okayama University HospitalBackground Postoperative osimertinib for EGFR mutant non-small cell lung cancer has become the standard of care. However, its adverse events in clinical practice remain unclear. We report a case of interstitial lung disease and pulmonary embolism occurring simultaneously as adverse events during adjuvant osimertinib treatment.<br>
Case presentation A 74-year-old woman, diagnosed with left lower lobe lung adenocarcinoma harboring an EGFR mutation, underwent a left lower lobectomy with lymph node dissection. During adjuvant osimertinib therapy, the patient developed respiratory distress with hypoxia, leading to the diagnosis of interstitial lung disease. Despite immediate steroid therapy, respiratory distress persisted, the patient developed leg edema. She was diagnosed with deep vein thrombosis and pulmonary embolism via contrast-enhanced computed tomography scan. Following treatment with steroid and anticoagulation, her clinical symptoms improved rapidly, and she showed no recurrence of interstitial lung disease, pulmonary embolism, or lung cancer over the following nine months.<br>
Conclusions We encountered a case of interstitial lung disease and pulmonary embolism occurring simultaneously as adverse events during adjuvant osimertinib treatment. In patients with osimertinib-induced interstitial lung disease, particularly when respiratory symptoms show poor improvement with steroid treatment, the possibility of pulmonary embolism complications should be suspected.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1868-43002025Efficacy of transcatheter patent foramen ovale closure for drug-resistant migraine: initial experience in Japan and long-term outcomeENTeijiAkagiDepartment of Cardiovascular Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYoichiTakayaDepartment of Cardiovascular Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTakashiMikiDepartment of Cardiovascular Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityRieNakayamaDepartment of Cardiovascular Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityKojiNakagawaDepartment of Cardiovascular Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityMitsukiNakashimaDepartment of Cardiovascular Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYoshiakiTakahashiDepartment of Neurology, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityNozomiHishikawaDepartment of Neurology, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityShinsukeYuasaDepartment of Cardiovascular Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityThis study evaluates the efficacy and safety of transcatheter patent foramen ovale (PFO) closure for the treatment of drug-resistant migraine in Japan. Previous studies have suggested a potential benefit for migraine with aura, although large-scale trials in the United States and Europe have failed to confirm efficacy as a primary endpoint. The study included 27 patients (mean age 36.4 years, 15 female, 21 with aura) who had more than two migraine attacks per month despite medication. All had PFO confirmed by transesophageal echocardiography and underwent transcatheter closure with the Amplatzer PFO Occluder. Patients were followed up to 12 months with migraine severity monitored by headache specialist. The procedure was successful and without complications in all cases. One patient required a larger occluder (35 mm) due to the size of PFO. At 12 months, 22 of 27 (81%) patients reported either complete resolution or improvement of migraine. Specifically, 10 of 21 (48%) patients with aura experienced complete resolution of migraine at one year. Patients without aura had a lower response rate, with only one case of complete resolution. Despite limitations such as the lack of a control group and potential patient selection bias, the study demonstrated that PFO closure may provide significant relief for patients with drug-resistant migraine, particularly those with aura. These findings support further investigation to better define its clinical indications and potential benefits.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2378-80381012025Short‐process incudo‐stapedioplasty in congenital ear malformatione70055ENRyotaroOmichiDepartment of Otolaryngology-Head and Neck Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShinKariyaDepartment of Otolaryngology-Head and Neck Surgery, Kawasaki Medical SchoolAkikoSugayaDepartment of Otolaryngology-Head and Neck Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineMizuoAndoDepartment of Otolaryngology-Head and Neck Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineObjectives: Although various stapedotomy and stapedectomy techniques exist, anchoring the piston can be challenging. We present a novel surgical approach for treating congenital stapes malformations with an atypical facial nerve trajectory.<br>
Methods: This is a case of a 7-year-old boy presenting with bilateral conductive hearing loss. Prior attempts at tympanoplasty had proven unsuccessful in improving his hearing. Presurgical imaging studies revealed an unusual anatomical configuration, with the facial nerve positioned inferior to the oval window. This anatomical variation precluded the use of conventional prosthesis-anchoring techniques typically employed in stapedotomies. Thus, we devised an innovative approach, opting to anchor the prosthesis to the short process of the incus.<br>
Results: This novel technique circumvented the atypical course of the facial nerve, allowing for successful reconstruction of the ossicular chain. The patient demonstrated an acceptable improvement (30 dB gain) in hearing 1-year post-surgery, with no reported complications.<br>
Conclusion: This case underscores the critical importance of adapting surgical techniques to address the unique anatomical challenges that may arise in the context of congenital ear malformations. It also highlights the potential of the short process of the incus as a viable alternative anchoring site for stapes prostheses, thereby improving the outcomes of such complex cases. This technique not only restored the patient's hearing but also contributed valuable insights into the management of similar cases, potentially improving the quality of life for individuals with rare and challenging anatomical variations.<br>
Level of evidence: 5.No potential conflict of interest relevant to this article was reported.BMJActa Medica Okayama2632-6140712025Reversible cerebral vasoconstriction syndrome in idiopathic multicentric Castleman disease under treatment with tocilizumabe000923ENNaoyaKamimuraDepartment of Neurology, Yokohama City University Medical CenterNaohisaUedaDepartment of Neurology, Yokohama City University Medical CenterKatsuoKimuraDepartment of Neurology, Yokohama City University Medical CenterAsamiNishikoriYasuharuSatoHitaruKishidaDepartment of Neurology, Yokohama City University Medical CenterFumiakiTanakaDepartment of Neurology and Stroke Medicine, Yokohama City University Graduate School of MedicineBackground Idiopathic multicentric Castleman disease (iMCD) is a rare polyclonal lymphoproliferative disorder characterised by systemic inflammation resulting from overproduction of interleukin 6 (IL-6). While iMCD primarily affects the lymph nodes and related tissues, it can also rarely involve the central nervous system.<br>
Case presentation We report the case of a 58-year-old female patient with at least a 3-year history of iMCD, who experienced acute thunderclap headaches due to reversible cerebral vasoconstriction syndrome (RCVS). RCVS occurred 3 months after initiating treatment with tocilizumab, a humanised anti-IL-6 receptor monoclonal antibody, and was accompanied by focal cortical subarachnoid haemorrhage (SAH). Elevated IL-6 levels were found in both serum and cerebrospinal fluid. MR angiography revealed multiple diffuse stenotic lesions in the bilateral middle and posterior cerebral arteries, which, along with bilateral cerebral oedema, resolved within 3 months. The diffuse nature of the cerebral vasospasm and the presence of bilateral brain oedema suggested that cerebral vasospasm was due to RCVS rather than SAH.<br>
Conclusions In patients with Castleman disease, RCVS may occur due to IL-6-dependent chronic cerebral vascular inflammation, either as a primary condition or as a complication of tocilizumab treatment.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841762025Managing Persistent Pupillary Membranes With Surgery or Medication: A Report of Three Casese86695ENToshihikoMatsuoDivision of Healthcare Science, Graduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityThe persistent pupillary membrane, as a congenital anomaly, is a remnant of a network of feeding blood vessels for the lens of the eye, called tunica vasculosa lentis. This study reports three patients with persistent pupillary membrane in both eyes who presented in different situations and were managed differently to achieve better vision. The first child (Case 1) who had been seen initially at the age of two years complained of severe photophobia even though he had good visual acuity, and hence, he and his family chose surgical resection of the pupillary membrane in both eyes at the age of six years just before the admission to an elementary school. He did not develop any surgical complications, such as cataract and glaucoma, and maintained the visual acuity in decimals of 1.2 in both eyes at the age of 17 years.<br>
The second child (Case 2), who was seen first at the age of one month, had persistent pupillary membranes in both eyes, together with Peters' anomaly in the left eye. The iris process adhesion to the corneal inner surface was visualized later by optical coherence tomography. She wore full-correction glasses and obtained the visual acuity of 0.7 in the right eye, so she had no problem studying at an elementary school. She used topical 1% atropine once a week in both eyes to maintain pupillary dilation and also used 0.5% timolol and 1% brinzolamide as pressure-lowering eye drops in the left eye with Peters' anomaly.<br>
The third patient (Case 3) with persistent pupillary membranes in both eyes complained of vision problems for the first time at the age of 49 years when she developed cataract. Surgical resection of the pupillary membrane was done in the initial phase of cataract surgery with intraocular lens implantation in both eyes. At surgical resection of the pupillary membrane, a safe and efficient way was to cut the root of the pupillary membrane on the iris surface with scissors, and then the isolated tissues of the pupillary membrane were pulled out with forceps from the side port at the corneal limbus. Pathological examinations of the excised tissues showed blood vessels with red blood cells in the lumen. In such a rare congenital disease as the persistent pupillary membrane, a case-based approach to choose a better option in different conditions from individual to individual is still required to have a better vision in learning at school and in daily working life.No potential conflict of interest relevant to this article was reported.Elmer Press, Inc.Acta Medica Okayama1923-41551652025Nephronophthisis and Retinitis Pigmentosa (Senior-Loken Syndrome) After Living-Donor Kidney Transplantation: Twelve-Year Follow-Up in a Young Woman164173ENToshihikoMatsuoGraduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityYasuhiroOnishiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityHiroshiMorinagaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityMotooArakiDepartment of Urology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversitySenior-Loken syndrome is a hereditary ciliopathy with recessive trait that manifests as nephronophthisis and retinitis pigmentosa. This report described an 18-year-old woman who was referred to a University Hospital to set up a treatment plan for chronic renal failure of an unknown cause. She had experienced nocturnal polyurea from the age of 12 years and was found to have an elevated level of serum creatinine at 3 mg/dL at the age of 15 years. She underwent renal biopsy at a hometown regional hospital which showed global glomerulosclerosis in six of the 13 glomeruli examined, renal tubular dilation in irregular shape, and marked interstitial fibrosis with lymphocytic infiltration. At the age of 19 years, she received a living-donor kidney transplant from her 46-year-old father as a preemptive therapy. At surgery, biopsy of the father’s donor kidney showed two glomeruli with global sclerosis out of 24 glomeruli examined, in association with minimal interstitial fibrosis and lymphocytic infiltration. She began to have extended-release tacrolimus 4 mg daily and mycophenolate mofetil 1,000 mg daily. According to the standard protocol, she underwent biopsy of the transplanted donor kidney to reveal interstitial fibrosis and lymphocytic infiltration, in addition to no sign of rejection and no glomerular deposition of immunoglobulins and complements, both 4 weeks and 14 months after the kidney transplantation. At the age of 23 years, 4 years after the kidney transplantation, she was, for the first time, diagnosed retinitis pigmentosa, and hence, Senior-Loken syndrome. She was followed up in the stable condition with basal doses of tacrolimus 5 mg daily, mycophenolate mofetil 1,000 mg daily, and prednisolone 5 mg daily up until now in 12 years after the kidney transplantation. The interstitial fibrosis with lymphocytic infiltration in the donor kidney might be a milder presentation of the disease with recessive inheritance.No potential conflict of interest relevant to this article was reported.Japanese Society of Internal MedicineActa Medica Okayama0918-29186452025A Novel De Novo Variant in KCNH5 in a Patient with Refractory Epileptic Encephalopathy759762ENAkihikoMitsutakeDepartment of Neurology, Graduate School of Medicine, The University of TokyoTakashiMatsukawaDepartment of Neurology, Graduate School of Medicine, The University of TokyoTatsuhikoNaitoDepartment of Neurology, Graduate School of Medicine, The University of TokyoHiroyukiIshiuraDepartment of Neurology, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityJunMitsuiDepartment of Neurology, Graduate School of Medicine, The University of TokyoHiroakiHaradaDepartment of Rheumatology and Allergy, Graduate School of Medicine, The University of TokyoKeishiFujioDepartment of Rheumatology and Allergy, Graduate School of Medicine, The University of TokyoJunFujishiroDepartment of Pediatric Surgery, Graduate School of Medicine, The University of TokyoHarushiMoriDepartment of Radiology, School of Medicine, Jichi Medical UniversityShinichiMorishitaDepartment of Computational Biology and Medical Sciences, Graduate School of Frontier Sciences, The University of TokyoShojiTsujiDepartment of Neurology, Graduate School of Medicine, The University of TokyoTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of TokyoWe herein report a novel de novo KCNH5 variant in a patient with refractory epileptic encephalopathy. The patient exhibited seizures at 1 year and 7 months old, which gradually worsened, leading to a bedridden status. Brain magnetic resonance imaging (MRI) showed cerebral atrophy and cerebellar hypoplasia. A trio whole-exome sequence analysis identified a de novo heterozygous c.640A>C, p.Lys214Gln variant in KCNH5 that was predicted to be deleterious. Recent studies have linked KCNH5 to various epileptic encephalopathies, with many patients showing normal MRI findings. The present case expands the clinical spectrum of the disease, as it is characterized by severe neurological prognosis, cerebral atrophy, and cerebellar hypoplasia.No potential conflict of interest relevant to this article was reported.SAGE PublicationsActa Medica Okayama2214-35992025Recent progress in oculopharyngodistal myopathy research from clinical and genetic viewpointsENHiroyukiIshiuraDepartment of Neurology, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityOculopharyngodistal myopathy (OPDM) is a rare muscular disorder characterized by ocular symptoms, pharyngeal symptoms, facial weakness, and distal predominant limb muscle weakness. The cause of the disease was unknown for a long time. Recently, however, it has been reported that expansions of CGG or CCG repeats in LRP12, LOC642361/NUTM2B-AS1, GIPC1, NOTCH2NLC, RILPL1, and ABCD3 are the causes of the disease. Cases sometimes present with neurological symptoms, and the clinical spectrum of diseases caused by expansions of CGG or CCG repeats has been proposed to be called FNOP-spectrum disorder after the names of fragile X-associated tremor/ataxia syndrome, neuronal intranuclear inclusion disease, oculopharyngeal myopathy with leukoencephalopathy, and OPDM. In this article, the recent progress in the field of OPDM is reviewed, and remaining issues in OPDM are discussed.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2054-345X1212025In-frame deletion variant of ABCD1 in a sporadic case of adrenoleukodystrophy5ENTakashiMatsukawaDepartment of Neurology, Graduate School of Medicine, The University of TokyoAtsushiSudoDepartment of Neurology, Graduate School of Medicine, The University of TokyoToshiyukiKakumotoDepartment of Neurology, Graduate School of Medicine, The University of TokyoAkihitoHaoDepartment of Neurology, Graduate School of Medicine, The University of TokyoMitsuhiroKainagaDepartment of Neurology, Graduate School of Medicine, The University of TokyoHyangriChangDepartment of Neurology, Graduate School of Medicine, The University of TokyoTatsuoManoDepartment of Neurology, Graduate School of Medicine, The University of TokyoHiroyukiIshiuraDepartment of Neurology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunMitsuiDepartment of Precision Medicine Neurology, Graduate School of Medicine, The University of TokyoToshihiroHayashiDepartment of Neurology, Graduate School of Medicine, The University of TokyoShinichiMorishitaDepartment of Computational Biology and Medical Sciences, Graduate School of Frontier Sciences, The University of TokyoShojiTsujiDepartment of Neurology, Graduate School of Medicine, The University of TokyoTatsushiTodaDepartment of Neurology, Graduate School of Medicine, The University of TokyoAdrenoleukodystrophy (ALD), an X-linked leukodystrophy caused by pathogenic variants in ABCD1, exhibits a broad range of phenotypes from childhood-onset cerebral forms to adult-onset adrenomyeloneuropathy (AMN). We report a rare in-frame ABCD1 deletion c.1469_71delTGG (p.Val490del) in a man with AMN. Although this variant has been interpreted as ‘uncertain significance’ in ClinVar, biochemical analysis along with clinical evaluation confirmed the pathogenicity of this variant, underscoring the importance of functional assessment of in-frame deletions.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama0893-39523882025The Involvement of PI3K–Akt Signaling in the Clinical and Pathological Findings of Idiopathic Multicentric Castleman Disease–Thrombocytopenia, Anasarca, Fever, Reticulin Fibrosis, and Organomegaly and Not Otherwise Specified Subtypes100782ENTomokaHaratakeDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesMidori FilizNishimuraDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesAsamiNishikoriDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesMichael V.GonzalezCenter for Cytokine Storm Treatment and Laboratory, Department of Medicine, Perelman School of Medicine, University of PennsylvaniaDaisukeEnnishiCenter for Comprehensive Genomic Medicine, Okayama University HospitalYou ChengLaiDepartment of Medical Biotechnology and Laboratory Science, Chang Gung UniversitySayakaOchiDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesManakaTsunodaDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesDavid C.FajgenbaumCenter for Cytokine Storm Treatment and Laboratory, Department of Medicine, Perelman School of Medicine, University of PennsylvaniaFritsvan RheeCenter for Cytokine Storm Treatment and Laboratory, Department of Medicine, Perelman School of Medicine, University of PennsylvaniaShujiMomoseDepartment of Pathology, Saitama Medical Center, Saitama Medical UniversityYasuharuSatoDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesIdiopathic multicentric Castleman disease is a rare lymphoproliferative disorder that is clinically classified into idiopathic plasmacytic lymphadenopathy (IPL); thrombocytopenia, anasarca, fever, reticulin fibrosis, and organomegaly (TAFRO); and not otherwise specified (NOS). Although each subtype shows varying degrees of hypervascularity, no statistical data on the degree of vascularization have been reported. Additionally, the mechanisms underlying vascularization in each clinical subtype are poorly understood. Here, we aimed to clarify these mechanisms by evaluating the histopathological characteristics of each clinical subtype across 37 patients and performing a whole-transcriptome analysis focusing on angiogenesis-related gene expression. Histologically, TAFRO and NOS exhibited a significantly higher degree of vascularization than IPL (IPL vs TAFRO, P < .001; IPL vs NOS, P = .002). In addition, the germinal centers (GCs) were significantly more atrophic in TAFRO than in IPL. In TAFRO and NOS, “whirlpool vessels” in GCs were seen in most cases (TAFRO, 9/9, 100%; NOS, 6/8, 75%) but not in IPL (IPL vs TAFRO, P < .001; IPL vs NOS, P = .007). Likewise, immunostaining for Ets-related gene revealed higher levels in endothelial cells of GCs in TAFRO than in IPL (P = .014), and TAFRO and NOS were associated with a significantly higher number of endothelial cells in interfollicular areas compared with that in IPL (TAFRO vs IPL, P < .001; NOS vs IPL, P = .002). Gene expression analysis revealed that the PI3K–Akt signaling pathway was significantly enriched in the TAFRO and NOS (TAFRO/NOS) groups. This pathway, which may be activated by vascular endothelial growth factor A and some integrins, is known to affect angiogenesis by increasing vascular permeability, which may explain the clinical manifestations of anasarca and/or fluid retention in TAFRO/NOS. These results suggest that the PI3K–Akt pathway plays an important role in the pathogenesis of TAFRO/NOS.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7932025A Case of Chromophobe Renal Cell Carcinoma Metastasizing to the Cervical Lymph Nodes after Long-term Follow-up213219ENMakotoWatanabeDepartment of Otolaryngology, Chugoku Rosai HospitalTomoyukiOgawaDepartment of Otolaryngology, Chugoku Rosai HospitalKanaoKobayashiDepartment of Nephrology and Urological Surgery, Chugoku Rosai HospitalNarutakaKatsuyaDepartment of Molecular Pathology, Graduate School of Medical Sciences, Hiroshima UniversityAkiraIshikawaDepartment of Molecular Pathology, Graduate School of Medical Sciences, Hiroshima UniversityTakaoHamamotoDepartment of Otolaryngology and Head and Neck Surgery, Hiroshima University HospitalHiroakiTaharaDepartment of Otolaryngology and Head and Neck Surgery, Hiroshima University HospitalTsutomuUedaDepartment of Otolaryngology and Head and Neck Surgery, Hiroshima University HospitalSachioTakenoDepartment of Otolaryngology and Head and Neck Surgery, Hiroshima University HospitalCase Report10.18926/AMO/68730Renal cell carcinoma (RCC) can metastasize hematogenously and recur after a long dormancy. Chromophobe RCC metastasized to the cervical lymph nodes 10 years after the primary resection in a woman who underwent nephrectomy for RCC (T1aN0M0 stage I). Metastatic RCC diagnosis was confirmed by aspiration. The lymph node mass was resected, and the tumor cells matched chromophobe RCC metastasis. No adjuvant therapy was administered due to the lack of evidence regarding adjuvant therapy for chromophobe RCC. Long-term surveillance is crucial in RCC because of the possibility of late metastasis. We reviewed the clinical aspects and literature on metastatic cervical RCC.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7932025A Case of Aniline Poisoning Manifesting as Cyanosis with Unknown Cause209212ENKenichiTaguchiDepartment of Gastroenterology, NHO Iwakuni Clinical CenterKazuyaNishiiDepartment of Respiratory Medicine, NHO Iwakuni Clinical CenterSakuraHataDepartment of Gastroenterology, NHO Iwakuni Clinical CenterShoichiKuyamaShoichiTanakaDepartment of Gastroenterology, NHO Iwakuni Clinical CenterCase Report10.18926/AMO/68729A 38-year-old man was brought to the hospital for emergency treatment of cyanosis. The patient exhibited generalized cyanosis and impaired consciousness despite adequate oxygen therapy. Arterial blood was black, and arterial blood gas analysis revealed an abnormally high methemoglobin level of 67.8%. We later interviewed his colleagues regarding his exposure to aniline while working at the factory and diagnosed him with methemoglobinemia due to aniline poisoning. The patient was administered methylene blue (MB) after being transferred to another hospital, where this treatment was available, resulting in an improvement in symptoms. Although rare, methemoglobinemia is serious. A good understanding of the circumstances at disease onset, characteristic findings, and abnormal values of methemoglobinemia is important. In addition, MB is an important therapeutic for the treatment of methemoglobinemia; if MB is not available at a particular hospital, transfer of the patient to a hospital that stocks MB should be considered.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7932025An Asymptomatic Perigraft Seroma in a Patient who Underwent Aortic Root Replacement for Annulo-Aortic Ectasia205208ENYasufumiFujitaDepartment of Cardiovascular Surgery, Kure Kyosai HospitalShujiShimizuDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/68728Perigraft seroma, a sterile fluid accumulation around the graft, is a potential complication after thoracic aortic surgery. The optimal treatment strategy for a perigraft seroma with vascular compression after thoracic aortic surgery has been unclear. We describe the case of a 62-year-old Japanese male in whom an asymptomatic perigraft seroma was observed after he had undergone aortic root replacement for annulo-aortic ectasia. The seroma was successfully treated with thoracoscopic drainage and conservative therapy. Less invasive therapy, including conservative therapy, may also be an option for asymptomatic perigraft seromas observed after thoracic aortic surgery.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7932025Rheumatoid Arthritis with Rapid Destructive Arthropathy of the Shoulder due to Calcium Pyrophosphate Deposition197203ENNaokiKondoDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesRikaKakutaniDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesTomoharuMochizukiDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesJunichi WakuiDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesNariakiHaoDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesEijiKinoshitaDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesHiroyukiKawashimaDivision of Orthopedic Surgery, Department of Regenerative and Transplant Medicine, Niigata University Graduate School of Medical and Dental SciencesCase Report10.18926/AMO/68727A 67-year-old woman with rheumatoid arthritis presented with an untriggered hematoma in the right shoulder joint. Radiographic findings showed humeral head collapse and destruction of the glenoid fossa with ectopic calcification. Calcium pyrophosphate deposition (CPPD) in the synovial fluid was observed using a polarizing microscope. Histopathological findings revealed chronic inflammatory cell infiltration and giant cells surrounded by CPPD. The patient was diagnosed with rapid destructive arthropathy (RDA). Endoscopic shoulder joint debridement was performed. Postoperatively, active flexion improved from 40 to 75 degrees. This case highlights that CPPD can cause RDA in the shoulder, detectable with detailed histopathology.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama0306-52512025Comprehensive analysis of adverse event profile changes with pertuzumab addition to trastuzumab‐based breast cancer therapy: Disproportionality analysis using VigiBaseENTatsuakiTakedaDepartment of Education and Research Center for Clinical Pharmacy, Faculty of Pharmaceutical Sciences, Okayama UniversityJunMatsumotoDepartment of Education and Research Center for Clinical Pharmacy, Faculty of Pharmaceutical Sciences, Okayama UniversityTomonoriSakaiDepartment of Pharmacy, Okayama University HospitalNaohiroIwataDepartment of Pharmacy, Okayama University HospitalHirofumiHamanoDepartment of Pharmacy, Okayama University HospitalToshihiroKoyamaDepartment of Health Data Science, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityNoritakaAriyoshiDepartment of Education and Research Center for Clinical Pharmacy, Faculty of Pharmaceutical Sciences, Okayama UniversityYoshitoZamamiDepartment of Pharmacy, Okayama University HospitalAims: Pertuzumab is used in combination with trastuzumab-based therapy for HER2-positive breast cancer. However, real-world safety information on pertuzumab remains limited. This study assessed the safety of adding pertuzumab to trastuzumab-based therapy for HER2-positive breast cancer using real-world data.<br>
Methods: VigiBase, the World Health Organization's global database of adverse events (AEs), containing reports from November 1967 to December 2023, was used. Signals for pertuzumab-associated AEs in breast cancer cases were detected using the reporting odds ratio (ROR).<br>
Results: Signals of trastuzumab plus pertuzumab relative to trastuzumab alone were detected in gastrointestinal disorders (ROR: 1.45, 95% confidence interval: 1.26–1.67), including diarrhoea (3.49, 2.83–4.30); infections and infestations (1.54, 1.24–1.91); and skin and subcutaneous tissue disorders (ROR: 1.63, 1.40–1.90), including pruritus (1.96, 1.51–2.55) and rash (1.63, 1.20–2.23). Further, signals of trastuzumab plus docetaxel plus pertuzumab relative to those of trastuzumab plus docetaxel were detected in gastrointestinal disorders (1.63, 1.38–1.93), including nausea (1.72, 1.24–2.39) and vomiting (1.48, 1.01–2.17), and in nervous system disorders (1.50, 1.20–1.87), including paraesthesia (2.60, 1.33–5.08) and peripheral sensory neuropathy (5.94, 1.79–19.71). The frequency of AEs causing or prolonging hospitalization was increased with trastuzumab plus pertuzumab compared to that with trastuzumab alone (1.18, 1.00–1.38).<br>
Conclusions: AE profiles after the addition of pertuzumab to trastuzumab-based therapy were comprehensively identified. The findings in this study highlight the importance of considering these AEs when selecting pertuzumab combination therapy to ensure the safety of patients with breast cancer.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama1744-99792932025Text mining for case report articles on “peritoneal dialysis” from PubMed database459470ENKazuhikoFukushimaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKenjiTsujiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHiroyukiNakanohDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesNaruhikoUchidaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSoichiroHaraguchiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinjiKitamuraDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesIntroduction: The number of published medical articles on peritoneal dialysis (PD) has been increasing, and efficiently selecting information from numerous articles can be difficult. In this study, we examined whether artificial intelligence (AI) text mining can be a good support for efficiently collecting PD information.<br>
Methods: We performed text mining and analyzed all the abstracts of case reports on PD in the PubMed database. In total, 3137 case reports with abstracts related to “peritoneal dialysis” published from 1970 to 2021 were identified.<br>
Results: A total of 280 347 relevant words were extracted from all the abstracts. Word frequency analysis, word dependency analysis, and word frequency transition analysis showed that peritonitis, encapsulating peritoneal sclerosis, and child have been important keywords. Theseanalyses not only reflected historical background but also anticipated future trends of PD study.<br>
Conclusion: These suggest that text mining can be a good support for efficiently collecting PD information.No potential conflict of interest relevant to this article was reported.S. Karger AGActa Medica Okayama0250-80952025Supplement-induced acute kidney injury reproduced in kidney organoidsENHiroyukiNakanohDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKenjiTsujiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKazuhikoFukushimaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesSoichiroHaraguchiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesShinjiKitamuraDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesIntroduction: Acute kidney injury associated with the consumption of Beni-koji CholesteHelp supplements, which contain red yeast rice (Beni-Koji), has become a significant public health concern in Japan. While renal biopsy findings from several case reports have suggested tubular damage, no definitive causal relationship has been established, and the underlying mechanisms of kidney injury remain poorly understood. The complexity of identifying toxic substances in supplements containing various bioactive compounds makes conventional investigative approaches both time-consuming and challenging. This highlights an urgent need to establish a reliable platform for assessing organ-specific toxicity in such supplements. In this study, we utilized a kidney organoid model derived from adult rat kidney stem cells (KS cells) to assess the potential tubular toxicity of these supplements. Methods: KS cell clusters were cultured in three-dimensional system supplemented with growth factors to promote kidney organoids. The organoids were subsequently exposed to Beni-koji CholesteHelp supplements or cisplatin, followed by histological and molecular analyses to evaluate structural impacts. Results: Established organoids had the kidney-like structures including tubular-like structures and glomerulus-like structures at the tips of multiple tubules. Treatment with Beni-koji CholesteHelp supplements induced significant tubular damage in the organoids, characterized by epithelial cell thinning, structural disruption, and increase in cleaved-caspase 3-positive apoptotic tubular cells, similar to the organoids treated with cisplatin. Conclusion: These findings provide the first evidence suggesting that certain toxicants in specific batches of Beni-koji CholesteHelp supplements cause direct renal tubular injury. This KS cell-based organoid system represents a cost-effective, reproducible, and technically simple platform for nephrotoxicity screening.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2769-2558422025Olanzapine enabled rechallenge after lorlatinib-induced psychosis: A case reporte70091ENAkiyoshiYokodeDepartment of Neuropsychiatry, Okayama University HospitalMasakiFujiwaraDepartment of Neuropsychiatry, Okayama University HospitalYukoNakamuraDepartment of Neuropsychiatry, Okayama University HospitalKadoakiOhashiDepartment of Respiratory Medicine, Okayama University HospitalShinjiSakamotoDepartment of Neuropsychiatry, Okayama University HospitalManabuTakakiDepartment of Neuropsychiatry, Okayama University Graduate School of Medicine,Dentistry, and Pharmaceutical SciencesBackground: Lorlatinib is a third-generation tyrosine kinase inhibitor for anaplastic lymphoma kinase (ALK)-positive non-small cell lung cancer (NSCLC). While it has a high intracranial lesion control rate, it can also cause central nervous system complications, including psychotic symptoms. We present a case of lorlatinib-induced psychosis successfully managed with olanzapine, enabling lorlatinib rechallenge.<br>
Case Presentation: A 32-year-old woman with ALK-positive NSCLC and brain metastases was started on lorlatinib. After 18 months, she developed hallucinations and delusions. Despite treatment with risperidone, her psychotic symptoms persisted, leading to hospitalization. Her symptoms resolved upon lorlatinib discontinuation while risperidone was continued. Given the critical role of lorlatinib in controlling brain metastases, rechallenge was considered. To mitigate concerns regarding drug interactions, risperidone was replaced with olanzapine. Following lorlatinib rechallenge with olanzapine, no recurrence of psychiatric symptoms was observed, allowing continued lorlatinib treatment. Additionally, no progression of lung cancer was noted.<br>
Conclusion: Lorlatinib is an essential drug for controlling brain metastases in ALK-positive NSCLC. However, it can induce psychotic symptoms. When psychiatrists are involved in managing adverse effects during cancer treatment, close collaboration among oncologists, psychiatrists, and patients is essential.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-09041342025A Case of a Metal Foreign Object Remaining in the Maxillary Bone for an Extended Period: A Case Reporte9631ENKoichiKadoyaDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYukiKunisadaDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKyoichiObataDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesHiroakiTakakuraDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTatsuoOgawaDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesSoichiroIbaragiDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesWe report a rare case in which a metallic foreign body remained undetected in a patient's maxilla for nearly 40 years after a childhood bicycle accident. Despite the accident, the implant remained in place without causing infection due to the lack of imaging studies at the time. The metal was accidentally discovered during a routine dental imaging examination 40 years later and subsequently surgically removed. This case highlights the importance of comprehensive imaging and the dangers of overlooking foreign bodies, especially in the vulnerable head and neck region. The patient, 53 years old at the time of discovery, presented to the dentist due to discomfort in the palate and nasal cavity. During this visit, radiographs were taken and a foreign body was discovered. Surgical removal of the foreign body revealed significant corrosion and surrounding granulation tissue indicative of foreign body granuloma. Elemental analysis of the foreign body confirmed that it was an iron-based metal, unlike biocompatible materials such as titanium. These findings reinforce the need for close post-trauma evaluation and follow-up, especially in cases of pediatric trauma, to avoid the possibility of long-term complications arising from unnoticed foreign bodies in anatomically significant areas.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2769-2558422025A case report of ineffective electroconvulsive therapy for chronic paine70108ENTakashiFukaoDepartment of Neuropsychiatry, Okayama University HospitalMasakiFujiwaraDepartment of Neuropsychiatry, Okayama University HospitalYutoYamadaDepartment of Neuropsychiatry, Okayama University HospitalKazushiAsadaDepartment of Neuropsychiatry, Okayama University HospitalTakahiroAsadaDepartment of Neuropsychiatry, Okayama University HospitalHirotoshiRiDepartment of Neuropsychiatry, Okayama University HospitalShinjiSakamotoDepartment of Neuropsychiatry, Okayama University HospitalManabuTakakiDepartment of Neuropsychiatry, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesBackground: Somatic symptom disorder (SSD), which includes chronic pain, is a common mental disorder characterized by significant functional impairment and other psychiatric comorbidities. Electroconvulsive therapy (ECT) has been proposed as a potential treatment for refractory chronic pain. However, evidence supporting its efficacy is limited and/or low quality. We present a case of SSD with chronic pain in which ECT was ineffective.<br>
Case Presentation: The patient was a 63-year-old man with chronic pain in the lower back, buttocks, thighs, and soles of the feet. The duration of his chronic pain was 3.8 years. He was diagnosed with Bertolotti's syndrome and SSD. He did not meet the criteria for major depressive disorder. He kept physically active by walking and doing exercises to distract himself from his pain. He strongly perceived pain as a physical issue and preferred ECT over psychotherapy. Despite undergoing 10 ECT sessions with adequate seizures, his pain persisted. After four sessions, he experienced despair over the lack of improvement in pain, which temporarily intensified his suicidal ideation. After undergoing ECT, he continued to maintain his activities, including walking and exercise, while his catastrophic thinking about pain persisted.<br>
Conclusion: The ineffectiveness of ECT in this case highlights the need for balanced counseling, particularly for patients who consider ECT a last-resort treatment. Psychological monitoring and depression screening are essential, especially given the risk of heightened despair or suicidal ideation when ECT is ineffective. Therefore, collaborative decision-making based on accurate information is vital.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7922025Safe Resection of Esophageal Cancer with a Non-Recurrent Inferior Laryngeal Nerve Associated with an Aberrant Right Subclavian Artery Using Intraoperative Nerve Monitoring139144ENYasushigeTakedaDepartment of Gastroenterological Surgery, Okayama University HospitalKazuhiroNomaDepartment of Gastroenterological Surgery, Okayama University HospitalNaoakiMaedaDepartment of Gastroenterological Surgery, Okayama University HospitalYoheiMizusawaDepartment of Gastroenterological Surgery, Okayama University HospitalHijiriMatsumotoDepartment of Gastroenterological Surgery, Okayama University HospitalYuheiKondoDepartment of Gastroenterological Surgery, Okayama University HospitalTomoyoshiKunitomoDepartment of Gastroenterological Surgery, Okayama University HospitalYukinoriTanoueDepartment of Gastroenterological Surgery, Okayama University HospitalMasashiHashimotoDepartment of Gastroenterological Surgery, Okayama University HospitalShunsukeTanabeDepartment of Gastroenterological Surgery, Okayama University HospitalToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University HospitalCase Report10.18926/AMO/68654In thoracic esophageal cancer, lymph node dissection around the recurrent laryngeal nerve is crucial but poses a risk of nerve palsy, affecting postoperative quality of life. In cases with an aberrant right subclavian artery (ARSA), the right recurrent laryngeal nerve is absent, and the non-recurrent inferior laryngeal nerve (NRILN) enters the larynx directly from the vagus nerve in the cervical region. Identifying the course of the NRILN is vital to avoid injury. A case of esophageal cancer with an ARSA, in which the course of the NRILN was preserved using the Nerve Integrity Monitoring (NIM) system during surgery, is described.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7922025Calcified Amorphous Tumor of the Left Ventricle with Paroxysmal Atrial Fibrillation135138ENYasufumiFujitaDepartment of Cardiovascular Surgery, Kure Kyosai HospitalShujiShimizuDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMakotoMohriDepartment of Cardiovascular Surgery, Japanese Red Cross Society Himeji HospitalCase Report10.18926/AMO/68653Cardiac calcified amorphous tumor (CAT) is a rare, benign non-neoplastic mass of the heart that is sometimes found due to embolic events. Most cases of CAT are treated with surgical removal to prevent future embolic events. However, the treatment strategy for CAT complicated by atrial fibrillation has remained to be determined. Here we report a case of left ventricular CAT complicated by paroxysmal atrial fibrillation (PAF) that was successfully treated with surgical removal and pulmonary vein isolation. Pulmonary vein isolation can be a simple and effective procedure for PAF, even during surgical removal of CAT.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7922025A Case of Retinitis Pigmentosa Diagnosed with Severe Anterior Capsule Contraction after Cataract Surgery129134ENAkihiroTsujiDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYusukeShiodeDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShuheiKimuraDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMioHosokawaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyoMatobaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTetsuroMoritaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKosukeTakahashiFukuyama City Hospital, Fukuyama CityYukiMorizaneDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/68652A 66-year-old woman presented with significant anterior capsule contraction and intraocular lens dislocation in both eyes 4 months after cataract surgery. Postoperative examinations such as fluorescein angiography, Goldmann perimetry, and electroretinography revealed retinitis pigmentosa (RP). Patients with significant anterior capsule contraction after cataract surgery should be closely examined because RP may be a contributing factor.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7922025Macular Hole Formation Six Months after Hemorrhage Displacement for Submacular and Henle Fiber Layer Hemorrhage due to Retinal Arterial Macroaneurysm Rupture123127ENRikuAkatsukaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShuheiKimuraDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyoMatobaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMioMorizane HosokawaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYusukeShiodeDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTetsuroMoritaDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinichiroDoiDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiMorizaneDepartment of Ophthalmology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/68651A 78-year-old woman presented with sudden vision loss and central scotoma. Visual acuity in the right eye was 20/222, with submacular hemorrhage (SMH) and Henle fiber layer hemorrhage (HFLh) due to retinal arterial macroaneurysm (RAM) rupture. She underwent SMH displacement, including cataract surgery, vitrectomy, intravitreal injection of tissue-plasminogen activator, and air tamponade. Three months postoperatively the SMH and HFLh had disappeared and visual acuity had improved to 20/200. Six months postoperatively, a macular hole had developed. We performed an inverted internal limiting membrane flap and gas tamponade. Ten months later, the hole had closed and visual acuity had improved to 20/100.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7922025From a Congenital Defect to Cancer: A Case of Squamous Cell Carcinoma in a Neglected Myelomeningocele117121ENAbhishekGautamDepartment of Neurosurgery, Jawaharlal Nehru Medical College, KAHERRahulKenawadekarDepartment of General Surgery, Jawaharlal Nehru Medical College, KAHERVirupaxiHattiholiDepartment of Radiology, Jawaharlal Nehru Medical College, KAHERPraful SureshMasteDepartment of Neurosurgery, Jawaharlal Nehru Medical College, KAHERCase Report10.18926/AMO/68650Neural tube defects are common congenital anomalies, typically presenting early due to visible swelling and/or neurological deficits. Rarely, cystic swellings are neglected until adulthood, with only 14 cases of malignancy developing in an untreated meningomyelocele reported to date. We describe the case details of a 26-year-old Indian woman with this rare complication. Magnetic resonance imaging revealed a low-lying spinal cord with spinal dysraphism, cord herniation, and a cystic lesion. The biopsy confirmed a well-differentiated squamous cell carcinoma. Malignant transformation in an untreated myelomeningocele is rare, with chronic irritation and infection as proposed causes. Early biopsy and treatment are crucial for its management.No potential conflict of interest relevant to this article was reported.Japanese Society for Pediatric EndocrinologyActa Medica Okayama0918-57393422025Effect of calcium supplementation on bone deformity and histopathological findings of skin papules in a pediatric patient with vitamin D–dependent rickets type 2A: A case report131136ENKoseiHasegawaDepartment of Pediatrics, Okayama University HospitalTomokoMiyakeDepartment of Dermatology, Okayama University HospitalMinaKobashiDepartment of Dermatology, Okayama University HospitalTomonoriTetsunagaDepartment of Dermatology, Okayama University HospitalYukoAgoDepartment of Pediatrics, Okayama University HospitalNatsukoFutagawaDepartment of Pediatrics, Okayama University HospitalHiroyukiMiyaharaDepartment of Pediatrics, Okayama University HospitalYousukeHiguchiDepartment of Pediatrics, Okayama University HospitalShinMorizaneDepartment of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHirokazuTsukaharaDepartment of Pediatrics, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesVitamin D–dependent rickets type 2A (VDDR2A) is an autosomal recessive disease caused by pathogenic variants of the vitamin D receptor (VDR) gene. VDDR2A rickets are usually resistant to native or active vitamin D treatment because of impaired active calcium absorption against the calcium concentration gradient, which is a ligand-dependent VDR action in the small intestine. Alopecia due to an impaired skin follicular cycle is occasionally observed in patients with VDDR2A. Among the pathogenic VDR variants, most in the DNA-binding domain and some in the ligand-binding domain, which affect the dimerization of VDR with the retinoic X receptor, are associated with alopecia. Herein, we report a case of VDDR2A caused by compound heterozygous pathogenic variants of the DNA-binding domain of VDR. Active vitamin D treatment did not ameliorate genu varum, rachitic changes in the roentgenogram, or abnormal laboratory findings. However, oral administration of calcium lactate dramatically improved these findings. The patient also experienced hair loss at two months of age and multiple papules on the skin at two yr of age, which did not improve with vitamin D or calcium supplementation. We also report the histopathological findings of skin papules in this patient.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841742025Bilateral Scleritis and Neutrophilic Dermatosis With Cytogenetic Chromosomal Aberrancy Related to Pyoderma Gangrenosum: A Case Report of a 20-Year Follow-Upe82348ENToshihikoMatsuoDepartment of Ophthalmology, Graduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityNoboruAsadaDepartment of Hematology and Oncology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityMikakoObikaDepartment of General Internal Medicine, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityRyotaroOmichiDepartment of Otolaryngology, Head and Neck Surgery, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityKeijiIwatsukiDepartment of Dermatology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityPyoderma gangrenosum is a non-infectious autoimmune disease with skin plaques and ulcers in the entity of neutrophilic dermatosis and may have a background of myelodysplastic syndromes. This study reported a 20-year follow-up of a patient with pyoderma gangrenosum and scleritis who showed chromosomal aberrancy from the initial phase and later in the course developed thrombocythemia. A 51-year-old man presented with widespread indurated erythematous plaques with scaling and pustules on the forehead, bilateral eyelids, and nasal bridge, in addition to nodular scleritis in the left eye and ulcer formation of the plaques in the lower legs. Skin biopsy revealed massive dermal infiltration mainly with neutrophils in the absence of neutrophilic vasculitis. Suspected of myelodysplastic syndromes, bone marrow biopsy was normal, while chromosomal aberrancy, 46, XY, del (20) (q11q13.3), was detected. In the diagnosis of neutrophilic dermatosis, probably of pyoderma gangrenosum, he began to have oral prednisolone 20 mg daily and colchicine 1 mg daily, leading to the subsidence of skin lesions. Four months later, he developed nodular scleritis in the right eye and began to use topical 0.1% betamethasone in both eyes. He was stable with only prednisolone 12.5 mg daily until the age of 55.5 years, when he showed an increase of serum lactate dehydrogenase. The bone marrow aspirate disclosed neither blast cell increase nor atypical cells. The same chromosomal aberrancy was repeatedly detected. One year later, he developed breathing difficulty and underwent tracheostomy. Laryngeal lesion biopsy disclosed squamous cell papilloma with human papillomavirus-6. At 60 years old, he showed marginal corneal infiltration in the left eye, and at 61 years old, hypopyon in the right eye. Platelets tended to increase up to 1000 × 103/µL, and bone marrow examinations were recommended but refused by the patient. At the latest follow-up at 71 years old, he was ambulatory in health and stable with a tracheostomy cannula. In conclusion, pyoderma gangrenosum with scleritis occurred in an undetermined hematological malignancy with chromosomal aberrancy.No potential conflict of interest relevant to this article was reported.MDPIActa Medica Okayama1661-65962662025Involvement of a Novel Variant of FGFR1 Detected in an Adult Patient with Kallmann Syndrome in Regulation of Gonadal Steroidogenesis2713ENYoshiakiSoejimaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMarinaKawaguchiDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKoheiOguniDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKoichiroYamamotoDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYasuhiroNakanoDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMihoYasudaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazukiTokumasuDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKeigoUedaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKoseiHasegawaDepartment of Pediatrics, Okayama University HospitalNahokoIwataDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFumioOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFibroblast growth factor receptor 1 (FGFR1), also known as KAL2, is a tyrosine kinase receptor, and variants of FGFR1 have been detected in patients with Kallmann syndrome (KS), which is a congenital developmental disorder characterized by central hypogonadism and anosmia. Herein, we report an adult case of KS with a novel variant of FGFR1. A middle-aged male was referred for a compression fracture of a lumbar vertebra. It was shown that he had severe osteoporosis, anosmia, gynecomastia, and a past history of operations for cryptorchidism. Endocrine workup using pituitary and gonadal stimulation tests revealed the presence of both primary and central hypogonadism. Genetic testing revealed a novel variant of FGFR1 (c.2197_2199dup, p.Met733dup). To identify the pathogenicity of the novel variant and the clinical significance for the gonads, we investigated the effects of the FGFR1 variant on the downstream signaling of FGFR1 and gonadal steroidogenesis by using human steroidogenic granulosa cells. It was revealed that the transfection of the variant gene significantly impaired FGFR1 signaling, detected through the downregulation of SPRY2, compared with that of the case of the forced expression of wild-type FGFR1, and that the existence of the variant gene apparently altered the expression of key steroidogenic factors, including StAR and aromatase, in the gonad. The results suggested that the novel variant of FGFR1 detected in the patient with KS was linked to the impairment of FGFR1 signaling, as well as the alteration of gonadal steroidogenesis, leading to the pathogenesis of latent primary hypogonadism.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-81841732025A Natural Course From Primary Intraocular Lymphoma to Brain Lymphoma in Four Years According to Patient's Choicee81476ENToshihikoMatsuoDepartment of Ophthalmology, Graduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityJojiIshidaDepartment of Neurological Surgery, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityShotaroKondoDepartment of Internal Medicine, Kurashiki Municipal HospitalKen-ichiMatsuokaDepartment of Hematology and Oncology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityPrimary intraocular lymphoma or vitreoretinal lymphoma is a rare entity of diffuse large B-cell lymphoma that presents vitreous opacity and retinal and choroidal infiltration. Primary central nervous system lymphoma would occur previously, later, or concurrently with respect to primary intraocular lymphoma. This study reported a 72-year-old patient with a pathological diagnosis of primary intraocular lymphoma who developed central nervous system lymphoma four years later in the course of no treatment. She presented with a four-year history of blurred vision in both eyes after cataract surgeries. Three weeks previously, she underwent a vitrectomy in the left eye at a clinic, and measurements of the vitreous fluid showed a high level of interleukin-10 at 5739 pg/mL, in contrast with interleukin-6 at 142 pg/mL. Cytology of the vitreous fluid was class III on the Papanicolaou classification. Head magnetic resonance imaging detected nothing abnormal. She underwent vitrectomy in the right eye as a diagnostic procedure to show large cells in the vitreous which were positive for CD20 and Ki-67 and negative for CD3, leading to a pathological diagnosis of large B-cell lymphoma. Prophylactic chemotherapy with high-dose methotrexate was recommended as a therapeutic option, but she chose observation since she did not have any eye or systemic symptoms. In the follow-up every three months by an oncologist and an ophthalmologist, she did not have any symptoms, and serum levels of soluble interleukin-2 receptor were in the normal range at each visit. She was well for four years until the age of 76 years when she fell and hit her head, and an emergency head computed tomography scan showed a mass in the left occipital lobe. Magnetic resonance imaging demonstrated a well-defined circular mass in the left occipital lobe with a hyperintense signal in the T2-weighted fluid-attenuated inversion recovery (FLAIR) image and diffusion-weighted image. Fluorodeoxyglucose positron emission tomography showed no abnormal uptake systemically, except for the left occipital lesion. She underwent a brain biopsy by craniotomy to pathologically prove diffuse large B-cell lymphoma. She was recommended to receive first-line chemotherapy as the standard treatment but chose observation with no treatment and died of brain lymphoma nine months later. This case happened to illustrate a natural course of primary intraocular lymphoma which proceeded to central nervous system lymphoma four years later.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama1878-87501952025Utility of Surgical Simulation for Tubular Retractor Surgery Using Three-Dimensional Printed Intraventricular Tumor Models: Case Series123743ENRyoOmaeDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyuKimuraDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshihiroOtaniDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJunHarumaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomoyaSaijoDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJuntaroFujitaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShoheiNishigakiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyosukeIkemachiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShuichiroHiranoDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesJojiIshidaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKentaroFujiiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakaoYasuharaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShotaTanakaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesObjective: The utility of the tubular retractor for deep-seated tumors, including intraventricular tumors, has recently been reported. However, the surgical field’s depth and narrowness can lead to blind spots, and it is crucial to prevent damage to the cortex and white matter fibers in eloquent areas. Therefore, preoperative simulation is critical for tubular retractor surgery. In this study, we investigated the benefits of threedimensional (3D)-printed intraventricular tumor models for tubular retractor surgery.<br>
Methods: Nine patients with intraventricular central neurocytoma who underwent tubular retractor surgery at our institution between March 2013 and August 2023 were retrospectively reviewed. Fusion images and 3D-printed intraventricular tumor models were developed from preoperative computed tomography (CT) and magnetic resonance imaging (MRI). The puncture points of the tubular retractor were simulated using fusion images and 3D-printed intraventricular tumor models by 11 neurosurgeons (3 experts in brain tumors, 2 experts in areas other than brain tumors, and 6 residents). The dispersion of puncture points among 8 neurosurgeons (excluding brain tumor experts) was compared in each simulation model.<br>
Results: These cases were categorized into two groups based on the dispersion of puncture points simulated by fusion images. Puncture point dispersion was markedly smaller in all cases when using 3D-printed intraventricular tumor models compared to simulations solely based on fusion images.<br>
Conclusions: In intraventricular tumor surgery using a tubular retractor, 3D-printed intraventricular tumor models proved more beneficial in preoperative simulation compared to fusion images.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2692-4609512025Endoscopic ultrasonography-guided removal of a stent that had migrated into the pancreas post-pancreaticojejunostomy: A case reporte70096ENSatoshiKajitaniDepartment of Gastroenterology andHepatology, Okayama University HospitalKazuyukiMatsumotoDepartment of Gastroenterology andHepatology, Okayama University HospitalKentaroOkiDepartment of Gastroenterology andHepatology, Okayama University HospitalAkihiroMatsumiDepartment of Gastroenterology andHepatology, Okayama University HospitalKazuyaMiyamotoDepartment of Gastroenterology andHepatology, Okayama University HospitalYukiFujiiDepartment of Gastroenterology andHepatology, Okayama University HospitalDaisukeUchidaDepartment of Gastroenterology andHepatology, Okayama University HospitalKoichiroTsutsumiDepartment of Gastroenterology andHepatology, Okayama University HospitalShigeruHoriguchiDepartment of Gastroenterology andHepatology, Okayama University HospitalMotoyukiOtsukaDepartment of Gastroenterology andHepatology, Okayama University HospitalA 64-year-old woman had undergone subtotal stomach-preserving pancreaticoduodenectomy for locally advanced pancreatic head cancer. She had an uneventful postoperative course with no recurrence. However, approximately 18 months after surgery, she presented with recurrent abdominal pain. Although contrast-enhanced computed tomography abdominal radiographs showed internal stent migration to the residual pancreas, dilatation of the tail side of the pancreatic duct was observed. The impaired internal stent was considered to be the cause of the abdominal pain. An attempt to remove the stent via balloon-assisted endoscopy was unsuccessful as the pancreaticojejunostomy site could not be reached. Consequently, endoscopic ultrasonography-guided pancreatic duct drainage was performed, and a plastic stent was placed through the jejunal site to the stomach. Two months later, the endosonographically/endoscopic ultrasonography-guided created route was dilated, and an endoscopic introducer was inserted into the pancreatic duct. Biopsy forceps were advanced through the sheath, allowing the successful removal of the stent by direct grasping. The symptoms of the patient improved, and she was discharged without complications.No potential conflict of interest relevant to this article was reported.MDPIActa Medica Okayama2077-03831442025Natural Course and Long-Term Outcomes of Gastric Subepithelial Lesions: A Systematic Review1055ENMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHiroyukiOkadaDepartment of Internal Medicine, Japanese Red Cross Society Himeji HospitalMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesBackground/Objectives: Gastric subepithelial lesions (SELs) are often incidentally detected during endoscopic examinations, with most patients being asymptomatic and lesions measuring <20 mm. Despite their generally indolent nature, certain SELs, such as gastrointestinal stromal tumors, require resection. Current guidelines recommend periodic surveillance; however, the natural course and long-term outcomes of gastric SELs have not been sufficiently investigated. This systematic review aimed to synthesize evidence on the progression, growth rate, and risk factors associated with gastric SELs to inform clinical management strategies. Methods: A comprehensive search of PubMed was conducted for peer-reviewed studies published between January 2000 and November 2024. Eligible studies included original studies on the follow-up and progression of gastric SELs. Non-English articles, reviews, case reports, and unrelated topics were excluded. In total, 277 articles were screened, with 15 additional articles identified through manual screening. Ultimately, 41 articles were included in the analysis. The study protocol is registered in PROSPERO (CRD42024614865). Results: Large-scale studies reported low growth rates of 2.0-8.5% over 2.0-5.0 years, while smaller studies reported a broader range of growth rates of 5.4-28.4%. The factors contributing to these discrepancies include patient selection, follow-up duration, and growth criteria. Risk factors for lesion size increase include larger initial lesion size, irregular margins, heterogeneous echo patterns, and certain tumor locations. Conclusions: These findings underscore the need for individualized management strategies based on lesion size, imaging characteristics, and risk factors. The close monitoring of high-risk lesions is crucial for timely intervention. Standardized growth criteria and optimized follow-up protocols are essential for improving clinical decision making and patient outcomes.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2769-2558412025A case of invasive pulmonary aspergillosis associated with clozapine-induced agranulocytosise70077ENAkiyoshiYokodeDepartment of Neuropsychiatry, Okayama University HospitalMasakiFujiwaraDepartment of Neuropsychiatry, Okayama University HospitalToshikiTeraoDepartment of Hematology and Oncology, Okayama University HospitalShinjiSakamotoDepartment of Neuropsychiatry, Okayama University HospitalYutoYamadaDepartment of Neuropsychiatry, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesRyotaSatoOkayama Psychiatric Medical CenterMomokoMishimaOkayama Psychiatric Medical CenterYujiYadaOkayama Psychiatric Medical CenterKen-IchiMatsuokaDepartment of Hematology, Endocrinology and Metabolism, Tokushima University Graduate School of Biomedical SciencesManabuTakakiDepartment of Neuropsychiatry, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesBackground: Clozapine-induced agranulocytosis (CLIA) is a rare but serious complication. Fever associated with CLIA is typically treated with broad-spectrum antimicrobials, but empiric antifungal therapy is rarely used. While bacterial and viral infections have been reported in CLIA cases, no cases of fungal infections complicated by CLIA have been documented. We report the first case of CLIA complicated by invasive pulmonary aspergillosis (IPA) in a patient with schizophrenia. The diagnosis of IPA was made using serum beta-D-glucan, Aspergillus galactomannan antigen tests, and chest computed tomography (CT). <br>
Case presentation: We present a case of a 51-year-old man with schizophrenia who developed CLIA complicated by IPA. The patient, diagnosed with treatment-resistant schizophrenia, was started on clozapine, but 9 months later he presented with fever, cough, leukopenia, and neutropenia. Clozapine was discontinued, and empirical treatments with cefepime and filgrastim were initiated. Serum beta-D-glucan and Aspergillus galactomannan antigen tests were positive, and chest CT showed well-circumscribed nodules, leading to a probable diagnosis of IPA. Antifungal therapy was switched from micafungin to voriconazole according to guidelines. His neutropenia and fever improved, and he was re-transferred to a psychiatric hospital. <br>
Conclusion: CLIA can be complicated by fungal infections. When patients with CLIA present with fever, fungal infections, including IPA, should be considered in the differential diagnosis. Serological tests, including beta-D-glucan and Aspergillus galactomannan, are useful for the diagnosis of IPA as well as the appropriate use of antifungal agents in patients with CLIA.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama0361-860996102021Validated international definition of the thrombocytopenia, anasarca, fever, reticulin fibrosis, renal insufficiency, and organomegaly clinical subtype (TAFRO) of idiopathic multicentric Castleman disease12411252ENYoshitoNishimuraDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesDavid C.FajgenbaumCenter for Cytokine Storm Treatment & Laboratory, Division of Translational Medicine and Human Genetics, Perelman School of Medicine, University of PennsylvaniaSheila K.PiersonCenter for Cytokine Storm Treatment & Laboratory, Division of Translational Medicine and Human Genetics, Perelman School of Medicine, University of PennsylvaniaNorikoIwakiHematology/Respiratory Medicine, Kanazawa University Graduate School of Medical ScienceAsamiNishikoriDivision of Pathophysiology, Okayama University Graduate School of Health SciencesMitsuhiroKawanoDepartment of Rheumatology, Kanazawa University Graduate School of Medical ScienceNaoyaNakamuraDepartment of Pathology, Tokai University School of MedicineKojiIzutsuDepartment of Hematology, National Cancer Center HospitalKengoTakeuchiDepartment of Pathology, The Cancer Institute Hospital of Japanese Foundation for Cancer ResearchMidori FilizNishimuraDepartment of Pathology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYoshinobuMaedaDepartment of Hematology, Oncology and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFumioOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuyukiYoshizakiDepartment of Organic Fine Chemicals, Institute of Scientific and Industrial Research, Osaka UniversityEricOksenhendlerDepartment of Clinical Immunology, Hôpital Saint-LouisFritsvan RheeMyeloma Center, University of Arkansas for Medical SciencesYasuharuSatoDivision of Pathophysiology, Okayama University Graduate School of Health SciencesThrombocytopenia, anasarca, fever, reticulin fibrosis, renal insufficiency, and organomegaly (TAFRO) syndrome is a heterogeneous entity manifesting with a constellation of symptoms described above that can occur in the context of idiopathic multicentric Castleman disease (iMCD) as well as infectious diseases, malignancies, and rheumatologic disorders. So, iMCD-TAFRO is an aggressive subtype of iMCD with TAFRO syndrome and often hyper-vascularized lymph nodes. Since we proposed diagnostic criteria of iMCD-TAFRO in 2016, we have accumulated new insights on the disorder and additional cases have been reported worldwide. In this systematic review and cohort analysis, we established and validated a definition for iMCD-TAFRO. First, we searched PubMed and Japan Medical Abstracts Society databases using the keyword “TAFRO” to extract cases. Patients with possible systemic autoimmune diseases and hematologic malignancies were excluded. Our search identified 54 cases from 50 articles. We classified cases into three categories: (1) iMCD-TAFRO (TAFRO syndrome with lymph node histopathology consistent with iMCD), (2) possible iMCD-TAFRO (TAFRO syndrome with no lymph node biopsy performed and no other co-morbidities), and (3) TAFRO without iMCD or other co-morbidities (TAFRO syndrome with lymph node histopathology not consistent with iMCD or other comorbidities). Based on the findings, we propose an international definition requiring four clinical criteria (thrombocytopenia, anasarca, fever/hyperinflammatory status, organomegaly), renal dysfunction or characteristic bone marrow findings, and lymph node features consistent with iMCD. The definition was validated with an external cohort (the ACCELERATE Natural History Registry). The present international definition will facilitate a more precise and comprehensive approach to the diagnosis of iMCD-TAFRO.No potential conflict of interest relevant to this article was reported.Japanese Society for Lymphoreticular Tissue ResearchActa Medica Okayama1346-42806422024Atypical lymphoplasmacytic and immunoblastic proliferation: A Systematic Review97106ENMidori FilizNishimuraDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesToshiakiTakahashiDepartment of Medicine, John A. Burns School of Medicine, University of Hawai’iKensukeTakaokaDepartment of Medicine, John A. Burns School of Medicine, University of Hawai’iSharinaMacapagalDepartment of Medicine, John A. Burns School of Medicine, University of Hawai’iChalothornWannaphutDepartment of Medicine, John A. Burns School of Medicine, University of Hawai’iAsamiNishikoriDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesHirokoTodaDepartment of Pathology, Chugoku Central HospitalYoshitoNishimuraDepartment of Medicine, John A. Burns School of Medicine, University of Hawai’iYasuharuSatoDepartment of Molecular Hematopathology, Okayama University Graduate School of Health SciencesAtypical lymphoplasmacytic and immunoblastic proliferation (ALPIBP) was first reported in 1984 as characteristic histological findings in lymph nodes associated with autoimmune diseases, but it has not been clearly defined to date. To summarize the histological characteristics and clinical diagnoses associated with ALPIBP, we searched MEDLINE and EMBASE for all peer-reviewed articles using keywords including “atypical lymphoplasmacytic and immunoblastic lymphadenopathy” from their inception to December 27, 2023. We also summarized the courses of three cases with a pathological diagnosis of ALPIBP. Nine articles with 52 cases were included. Among the total of 55 cases, including the three from our institution, the median age of the cases was 63.5 years with a female predominance (69.5%). Lymphadenopathy was generalized in 65.6% and regional in 34.4% of cases. RA (24.4%), SLE (24.4%), and autoimmune hemolytic anemia (20.0%), were common clinical diagnoses. A combination of cytotoxic chemotherapy was used in 15.6% of cases due to the suspicion of malignancy. Nodal T-follicular helper cell lymphoma, angioimmunoblastic type, methotrexate-associated lymphoproliferative disorders, and IgG4-related diseases were listed as important diseases that need to be pathologically differentiated from ALPIBP. This review summarizes the current understanding of the characteristics of ALPIBP. Given that underrecognition of ALPIBP could lead to overdiagnosis of hematological malignancy and unnecessary treatment, increased awareness of the condition in pathologists and clinicians is crucial.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1865-72571822025A case of pancreatic ductal adenocarcinoma growing within the pancreatic duct mimicking an intraductal tubulopapillary neoplasm376382ENRyosukeSatoDepartment of Gastroenterology and Hepatology, Okayama University HospitalKazuyukiMatsumotoDepartment of Gastroenterology and Hepatology, Okayama University HospitalMayuUkaDepartment of Radiology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKoseiTakagiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKenjiNishidaDepartment of Pathology, Dentistry and Pharmaceutical Science, Okayama University Graduate School of MedicineTakehiroTanakaDepartment of Pathology, Dentistry and Pharmaceutical Science, Okayama University Graduate School of MedicineYukiFujiiDepartment of Gastroenterology and Hepatology, Okayama University HospitalKoichiroTsutsumiDepartment of Gastroenterology and Hepatology, Okayama University HospitalShigeruHoriguchiDepartment of Gastroenterology and Hepatology, Okayama University HospitalMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University HospitalWe herein report a case of pancreatic ductal adenocarcinoma (PDAC) that developed within the pancreatic duct and was initially diagnosed as an intraductal tubulopapillary neoplasm (ITPN). A 76-year-old man presented with weight loss and main pancreatic duct dilation. The imaging studies revealed a 30-mm hypovascular tumor within the main duct of the pancreatic head. An endoscopic examination with a biopsy revealed high-grade atypical epithelial cells with immunostaining patterns suggestive of ITPN. Following robot-assisted pancreaticoduodenectomy, postoperative pathology revealed conflicting features: nodular/cribriform infiltrations typical of ITPN and non-lobular replacement with scattered infiltrations characteristic of PDAC. A comprehensive genomic profiling test detected KRAS and TP53 mutations, leading to the final diagnosis of PDAC (fT3N1aM0, stage IIB). The patient received adjuvant S-1 chemotherapy and remained recurrence-free for 15 months post-surgery. This case highlights the diagnostic challenges of differentiating intraductal pancreatic tumors and demonstrates the utility of integrating genetic testing with conventional diagnostic modalities for an accurate diagnosis and appropriate treatment selection.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7912025A Rare Case of Compression Neuritis due to Intraorbital Arteriovenous Fistula (IOAVF) Mimicking Retrobulbar Optic Neuritis5964ENShunMinakawaDepartment of Ophthalmology, Japanese Red Cross Society Himeji HospitalMasayukiHiranoDepartment of Ophthalmology, Japanese Red Cross Society Himeji HospitalKazuyaTakahashiDepartment of Neurosurgery, Japanese Red Cross Society Himeji HospitalYutaImamuraDepartment of Ophthalmology, Japanese Red Cross Society Himeji HospitalTakashiWatanabeDepartment of Ophthalmology, Japanese Red Cross Society Himeji HospitalCase Report10.18926/AMO/68363Intraorbital arteriovenous fistulas (IOAVFs) are rare vascular abnormalities. We describe a case of an IOAVF featuring a direct shunt between the accessory meningeal artery and the superior ophthalmic artery. A 55-year-old woman presented with a 1-month history of visual impairment in her right eye, and magnetic resonance imaging (MRI) revealed optic neuritis-like findings. Steroid pulse therapy temporarily resolved visual impairment. However, 1 month later, she experienced decreased visual acuity, ocular conjunctival hyperemia, edema, and a pulsatile murmur. Contrast-enhanced MRI and digital subtraction angiography revealed compression optic neuropathy due to an IOAVF. Following successful treatment with transarterial embolization, her symptoms disappeared.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7912025Photoinitiators Induce Histamine Production in Human Mast Cells5158ENTaroMiuraDepartment of Clinical Pharmacy, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoichiKawasakiLaboratory of Clinical Pharmacology and Therapeutics, Kagawa School of Pharmaceutical Sciences, Tokushima Bunri UniversityHirofumiHamanoDepartment of Clinical Pharmacy, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshitoZamamiDepartment of Clinical Pharmacy, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesToshiakiSendoDepartment of Clinical Pharmacy, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesOriginal Article10.18926/AMO/68362Photoinitiators are used in the manufacture of many daily products, and may produce harmful effects due to their cytotoxicity. They have also been detected in human serum. Here, we investigated the histamine-producing effects in HMC-1 cells and the inflammatory cytokine release effects in RAW264 cells for four photoinitiators: 1-hydroxycyclohexyl phenyl ketone; 2-isopropylthioxanthone; methyl 2-benzoylbenzoate; and 2-methyl-4´-(methylthio)-2-morpholinopropiophenone. All four promoted histamine production in HMC-1 cells; however, they did not significantly affect the release of inflammatory cytokines in RAW264 cells. These findings suggest that these four photoinitiators induce inflammatory cytokine-independent histamine production, potentially contributing to histamine-mediated chronic inflammation in vitro.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-09041322025Crowned Dens Syndrome Triggered by Dental Treatmente70168ENSatoruMoritaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoheiMasudaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshiakiSoejimaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFumioOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCrowned dens syndrome (CDS) is an important yet often overlooked cause of fever and neck pain, frequently leading to unnecessary examinations and treatments and misdiagnosis as infectious diseases or rheumatic diseases. The mechanism of an acute attack of CDS is not clarified completely, while it is considered that severe systemic stress can trigger inflammation caused by calcium pyrophosphate crystals. We describe a case of CDS triggered by localized physical stress to the neck and emphasize the importance of considering this condition in cases of fever and neck pain following dental treatment.No potential conflict of interest relevant to this article was reported.Japanese Society of Internal MedicineActa Medica Okayama0918-29182025Dyspnea with Hemidiaphragm Elevation in a Patient with Giant Cell Arteritis: A Case Report4055-24ENYosukeAsanoDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesYoshinoriMatsumotoDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesNatsukiKubotaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesYuyaTerajimaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKazuyaMatsumotoDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKentaShidaharaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKeiHiroseDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesTakatoNakadoiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesShoichiNawachiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesYuKatayamaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesYoshiaMiyawakiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesEriKatsuyamaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesTakayukiKatsuyamaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesMarikoTakano-NarazakiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesKen-EiSadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesWe herein report the first case of dyspnea with hemidiaphragm elevation in a 68-year-old woman with active giant cell arteritis (GCA), including successful treatment. Contrast-enhanced computed tomography showed a reduced density of the left ophthalmic artery and the left superficial temporal artery with increased soft tissue compared to the other side, indicating that the GCA had flared up and suggesting that the hemidiaphragm elevation might be caused by vasculitis-associated ischemia of the right phrenic nerve. Hemidiaphragm paralysis due to vasculitis-associated ischemia in patients with GCA needs to be distinguished from local infection, tumors, and hepatomegaly, which are the major causes of hemidiaphragm elevation.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2090-6447202512025A Case of an Oral Elastofibromatous Lesion: A Clinicopathological Analysis With a Literature Review5556176ENSawakoOnoDepartment of Pathology and Oncology, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityMasanoriMasuiDepartment of Oral and Maxillofacial Surgery, Kagawa Prefectural Central HospitalKyoichiObataDepartment of Oral and Maxillofacial Surgery, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTomoyaNakamuraDepartment of Oral and Maxillofacial Surgery, Kagawa Prefectural Central HospitalYoshihikoFurukiDepartment of Oral and Maxillofacial Surgery, Kagawa Prefectural Central HospitalSatokoNakamuraDepartment of Pathology, Kagawa Prefectural Central HospitalHidetakaYamamotoDepartment of Pathology and Oncology, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityElastofibromatous changes of the oral mucosa, such as an elastofibroma (EF) or an elastofibromatous lesion (EFL), are not well recognized, and the second such case in Japan is reported. A 72-year-old man wearing a complete maxillary denture presented with a small nodule on the hard palate. Histopathological examination showed abundant fibrous tissue with numerous elastic fibers on Elastica van Gieson (EvG) staining. The diagnosis of an oral EFL was made. In the review of oral EF and EFL, no cases with recurrence were identified, but such lesions may resemble neoplastic lesions macroscopically. Accurate diagnosis using EvG stain is needed to recognize oral EFs and EFLs.No potential conflict of interest relevant to this article was reported.BMCActa Medica Okayama2059-13811012024Case series showing the safety and changes in lipid profiles of hemodialysis patients with hypertriglyceridemia after pemafibrate administration74ENRinoOkadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesYasuhiroOnishiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesNaoyaKobayashiOkayama Saidaiji HospitalHiroyukiIshiharaOkayama Saidaiji HospitalTomohisaYokoyamaOkayama Saidaiji HospitalTomoyoMifuneOkayama Saidaiji HospitalYoshimasaSakurabuDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesIchiroNojimaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesHiroshiMorinagaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesHaruhito A.UchidaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesJunWadaDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Faculty of Medicine, Dentistry and Pharmaceutical SciencesBackground Cardiovascular disease is a major cause of morbidity and mortality in patients with chronic kidney disease and end-stage renal disease (ESRD). Dyslipidemia is a key focus of cardiovascular therapy and is characterized by hypertriglyceridemia mainly caused by lipoprotein lipase-mediated metabolism of ApoC-III in patients with ESRD. Pemafbrate, a selective peroxisome proliferator-activated receptor alpha modulator, can be used regardless of renal function and inhibit ApoC-III expression in the liver.<br>
Case presentation We reported the cases of four patients on hemodialysis who met at least 175 mg/dL of triglycerides on the consecutive three tests between September 2022 and November 2022 and took 0.1 mg pemafbrate twice a day from November 2022 to May 2023. They experienced no adverse events after pemafbrate treatment. Pemafbrate signifcantly reduced triglyceride (TG) (302±72 to 140±50 mg/dL, p=0.048), total cholesterol (187±34 to 156±48 mg/dL, p=0.025), and Apo C-III (15.9±8.2 to 12.6±7.1, p=0.030) levels. Apo A-II levels signifcantly increased after treatment (27.0±6.1 to 37.1±5.8, p=0.041).<br>
Conclusions Pemafbrate decreased TG, total cholesterol, and Apo-CIII and increased Apo A-II without adverse events. Further study is needed to examine the favorable efects of pemafbrate on the risk of CVD.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-09041312025Undetermined Ruptured Low-Grade Appendiceal Mucinous Neoplasm Following High-Energy Blunt Abdominal Trauma Requiring Emergency Laparotomye70071ENIppeiMatsuoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTetsuyaYumotoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityAkariTsujiDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityRyoTanabeDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityToshihisaMatsumuraDepartment of Digestive Surgery, Japanese Red Cross Okayama HospitalMikotoShimabaraDepartment of Digestive Surgery, Japanese Red Cross Okayama HospitalMasaakiAkaiDepartment of Digestive Surgery, Japanese Red Cross Okayama HospitalShojiTakagiDepartment of Digestive Surgery, Japanese Red Cross Okayama HospitalHiromichiNaitoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityAtsunoriNakaoDepartment of Emergency, Critical Care, and Disaster Medicine, Faculty of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityBlunt abdominal trauma causing intraperitoneal injury and/or bleeding can be life-threatening, requiring immediate intervention. Diagnosing these cases can be challenging, especially when pre-existing conditions are involved. Low-grade appendiceal mucinous neoplasm (LAMN) is a rare tumor of the appendix that can lead to pseudomyxoma peritonei. Herein, we present a case of ruptured LAMN following blunt abdominal trauma after a high-energy head-on collision, complicating the differentiation from other intraperitoneal injuries. A 42-year-old Japanese female was brought to our hospital following high-energy head-on collision. She presented with stable vital signs, complaining of anterior chest pain and abdominal tenderness without peritoneal irritation. Computed tomography scans indicated multiple fractures in her chest and complex fluid around the Douglas fossa extending to the ileocecal area, with a slightly dilated appendix tip. Despite stable vitals, emergency laparotomy was needed for suspected peritonitis and/or intraperitoneal hemorrhage. Emergency laparotomy revealed yellowish, jelly-like ascites and a ruptured appendiceal tumor. LAMN was suspected, and the appendix was completely resected, with cytoreductive surgery carefully performed. Histopathological examination confirmed the diagnosis of LAMN. Postoperative course was uneventful, and the patient was discharged on Day 13 and referred for further LAMN management. This case report highlights the diagnostic difficulties of LAMN rupture following blunt abdominal trauma, stressing the need to consider rare conditions like LAMN in differential diagnoses of acute abdomen posttrauma.No potential conflict of interest relevant to this article was reported.BMCActa Medica Okayama1880-65467412024Evaluation of the effects of fenestration in Fontan circulation using a lumped parameter model58ENNaohiroHorioDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShujiShimizuDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYasuhiroKotaniDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshinoriMiyaharaPediatric Heart Disease and Adult Congenital Heart Disease Center, Showa University HospitalShingoKasaharaDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFenestration has been reported to enhance Fontan hemodynamics in several cases of Fontan circulation. However, the indication criteria for fenestration remain under discussion. To assess the effectiveness of fenestration in Fontan circulation, we conducted a theoretical analysis using a computational model of the fenestrated Fontan circulation. The cardiac chambers and vascular systems were modeled using the time-varying elastance model and the modified Windkessel model, respectively. When the pulmonary vascular resistance index was 4.01 Wood units m2, fenestration significantly reduced central venous pressure from 18.0 to 16.1 mmHg and decreased stressed blood volume from 610 to 555 ml. However, in the models with reduced ventricular end-systolic elastance, increased ventricular stiffness constant, or heightened systemic vascular resistance, the advantages of fenestration were diminished. Thus, fenestration may effectively improve the hemodynamics of Fontan circulation in patients with elevated pulmonary vascular resistance.No potential conflict of interest relevant to this article was reported.ElsevierActa Medica Okayama1341-321X3122024Clinical and microbiological characteristics of high-level daptomycin-resistant Corynebacterium species: A systematic scoping review102575ENShinnosukeFukushimaDepartment of Bacteriology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalKazuyoshiGotohDepartment of Medical Laboratory Science, Okayama University Graduate School of Health SciencesShumaTsujiDepartment of Medical Laboratory Science, Okayama University Graduate School of Health SciencesKojiIioMicrobiology Division, Clinical Laboratory, Okayama University HospitalHidemasaAkazawaDepartment of Infectious Diseases, Okayama University HospitalOsamuMatsushitaDepartment of Bacteriology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesFumioOtsukaDepartment of Infectious Diseases, Okayama University HospitalIntroduction: Corynebacterium species potentially develop high-level daptomycin resistance (HLDR) shortly after daptomycin (DAP) administration. We aimed to investigate the clinical and microbiological characteristics of HLDR Corynebacterium infections.<br>
Methods: We first presented a clinical case accompanied by the results of a comprehensive genetic analysis of the isolate, and then performed a systematic scoping review. Based on the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Extension for Scoping Reviews, we searched for articles with related keywords, including “Corynebacterium”, “Daptomycin", and "Resistance”, in the MEDLINE and Web of Science databases from the database inception to October 25, 2024. Clinical case reports and research articles documenting the isolation of HLDR Corynebacterium species, defined by a minimum inhibitory concentration of DAP at ≥256 μg/mL, were deemed eligible for this review.<br>
Results: Of 80 articles screened, seven case reports detailing eight cases of HLDR Corynebacterium infections, as well as five research articles, were included. C. striatum was the most common species (7/9 cases, 77.8 %), and prosthetic device-associated infections accounted for 66.7 % of the cases. Duration of DAP administration before the emergence of HLDR isolates ranged from 5 days to 3 months; three-quarters of the cases developed within 17 days. Three HLDR isolates were genetically confirmed to have an alteration in pgsA2. The majority of the patients were treated with either glycopeptides or linezolid, with favorable outcomes. In vitro experiments confirmed that C. striatum strains acquire the HLDR phenotype at higher rates (71 %–100 %) within 24 h of incubation, compared to other Corynebacterium strains.<br>
Conclusion: DAP monotherapy, especially for prosthetic device-associated infections, can result in the development of HLDR Corynebacterium. Additional research is warranted to investigate the clinical implications of this potentially proliferating antimicrobial resistant pathogen.No potential conflict of interest relevant to this article was reported.ElsevierActa Medica Okayama1341-321X3112025Cryptococcal prostatitis in an immunocompromised patient with tocilizumab and glucocorticoid therapy: A case report102494ENKoheiOguniDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinnosukeFukushimaDepartment of Bacteriology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalAtsushiKatoDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAtsuhitoSuyamaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakehiroIwataDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshiaMiyawakiDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSawakoOnoDepartment of Pathology and Oncology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKojiIioMicrobiology Division, Clinical Laboratory, Okayama University HospitalFumioOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCryptococcus prostatitis is an uncommon manifestation of cryptococcal infection that occurs mostly in immunocompromised patients. Tocilizumab, an anti-interleukin-6 receptor monoclonal antibody, has been associated with an increased risk of cryptococcal infections. However, there have been no documented cases of cryptococcal prostatitis in patients receiving tocilizumab therapy. We report a case of cryptococcal prostatitis in a 72-year-old man treated with glucocorticoids and tocilizumab for giant cell arteritis and granulomatosis with polyangiitis. The patient presented dysuria and his serum level of prostate-specific antigen was elevated. Magnetic resonance imaging revealed a prostate mass, and a prostate biopsy was performed, leading to a pathologic diagnosis of cryptococcal prostatitis. Fungal cultures for blood and urine were negative, while the cryptococcal antigen for both serum and urine showed positive results. There were no particular findings in the pulmonary and central nervous systems. The patient was successfully treated with oral fluconazole (400 mg/day) and was discharged. Although cryptococcal prostatitis is a rare entity, clinicians should note that an immunosuppressed patient may develop such a difficult-to-diagnose disease.
No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2050-09041312025Eyelid Spindle Cell Lipoma: Case Report and Review of Three Patients in Literaturee70097ENToshihikoMatsuoGraduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityKiyoshiYamadaPlastic and Reconstructive Surgery, Kousei HospitalYasumasaMonobeDepartment of Pathology, General Medical Center, Kawasaki Medical SchoolTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityA 39-year-old woman presented a saucer-shaped mass in the left upper eyelid and underwent the extirpation at local anesthesia. Pathologically, collagen fibers, capillaries, small vessels, and CD34-positive spindle cells were dispersed among mature adipose tissues, indicative of spindle cell lipoma. Long-lasting cyst-like eyelid masses would be usually dermoid cysts, and spindle cell lipoma would be listed as a rare pathological diagnosis in differential diagnoses of cyst-like lesions in the upper and lower eyelid.No potential conflict of interest relevant to this article was reported.ElsevierActa Medica Okayama2210-26121262025Laparoscopic resection for oesophageal duplication cyst: A case report110572ENTomohiroHamazakiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKazuhiroNomaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKentoKawasakiDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasashiHashimotoDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShunsukeTanabeDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesIntroduction: Oesophageal duplication cyst is a congenital malformation and rare tumour, clinically manifesting as dysphagia, epigastric pain, or respiratory distress. Duplicate cysts associated with abscess formation or mediastinal penetration and malignancies have been reported, necessitating surgical resection. <br>
Presentation of case: A 55-year-old woman had chest discomfort for 1 year. Preoperative imaging, including computed tomography (CT), upper gastrointestinal endoscopy, and endoscopic ultrasound, revealed a tumour extending from the anterior wall to the lesser curvature of the near the oesophagogastric junction (OGJ) and a suspected mural nodule within the tumour. Contrast-enhanced CT revealed a cystic nodule on the wall of the lesser curvature of the OGJ, with an unclear boundary between the cystic nodule and the oesophageal wall. Magnetic resonance imaging showed an isointense signal on T1-weighted imaging and hyperintensity on T2weighted imaging. Laparoscopic lower oesophagectomy and proximal gastrectomy with lymph node dissection were performed to the confirm mucinous cyst. Pathological findings revealed a cystic lesion in the muscularis propria of the OGJ filled with mucinous components and lined with multilayered columnar epithelial cells. The cyst was diagnosed as a duplicate without malignancy. <br>
Discussion: Since the border between the cyst and the oesophageal walls was unclear, and the cyst potentially contained a malignant component, instead of cystectomy, lower oesophagectomy and proximal gastrectomy with lymph node dissection were performed with oesophagogastric anastomosis using the double-flap technique, tailored specifically for OGJ cancer. <br>
Conclusions: Oesophageal duplication cysts are rare. Lower oesophagectomy and proximal gastrectomy are selective surgical approaches for cyst duplication at the OGJ.No potential conflict of interest relevant to this article was reported.Elmer Press, Inc.Acta Medica Okayama1923-41551612025Local Control of Conjunctival Malignant Melanoma by Proton Beam Therapy in a Patient With No Metastasis in Six Years From in Situ to Nodular Lesions2836ENToshihikoMatsuoRegenerative and Reconstructive Medicine (Ophthalmology), Graduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityTakeshiOgataDepartment of Radiology, Proton Beam Center, Tsuyama Chuo HospitalTakahiroWakiDepartment of Radiology, Proton Beam Center, Tsuyama Chuo HospitalTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityKotaTachibanaDepartment of Dermatology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTomokazuFujiDepartment of Gastroenterological Surgery, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTakuyaAdachiDepartment of Gastroenterology and Hepatology, Okayama University HospitalOsamuYamasakiDepartment of Dermatology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityConjunctival malignant melanoma is extremely rare, with no standard of care established at moment. Here we report a 65-year-old woman, as a hepatitis B virus (HBV) carrier, who presented concurrently a liver mass and lower bulbar conjunctival pigmented lesions in the right eye. Needle liver biopsy and excisional conjunctival biopsy showed hepatocellular carcinoma and conjunctival malignant melanoma in situ, respectively. The priority was given to segmental liver resection for hepatocellular carcinoma after transcatheter arterial chemoembolization. In 1 year, she underwent second and third resection of bulbar conjunctival pigmented lesions, and the pathological examinations constantly showed melanoma in situ. In the course, she showed gradual widening of pigmented lesions to upper bulbar conjunctiva and lower palpebral conjunctiva and lower eyelid. About 2.5 years from the initial visit, the lower eyelid lesion was resected for a genomic DNA-based test of BRAF mutations which turned out to be absent, and then, she began to have intravenous anti-programmed cell death-1 (PD-1), nivolumab every 3 or 4 weeks. She developed iritis in the right eye with conjunctival melanoma as an immune-related adverse event, 3 months after the beginning of nivolumab, and so she used daily topical 0.1% betamethasone eye drops to control the intraocular inflammation. She showed no metastasis in 6 years of follow-up, but later in the course, 5 years from the initial visit, she developed abruptly a non-pigmented nodular lesion on the temporal side of the bulbar conjunctiva along the corneal limbus, accompanied by two pigmented nodular lesions in the upper and lower eyelids in a few months. She thus, underwent proton beam therapy toward the conjunctival melanoma and achieved the successful local control. Proton beam therapy is a treatment option in place of orbital exenteration, and multidisciplinary team collaboration is desirable to achieve better cosmetic and functional outcomes in conjunctival malignant melanoma.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7862024C-arm Free Unilateral Biportal Endoscopic Discectomy: A Technical Note475483ENHongfeiXiangDepartment of Orthopaedic Surgery, Okayama Rosai HospitalKajetanLatkaDepartment of Orthopaedic Surgery, Okayama Rosai HospitalPrafulMasteDepartment of Orthopaedic Surgery, Okayama Rosai HospitalMasatoTanakaDepartment of Orthopaedic Surgery, Okayama Rosai HospitalChetanKumawatDepartment of Orthopaedic Surgery, Okayama Rosai HospitalShinyaAratakiDepartment of Orthopaedic Surgery, Okayama Rosai HospitalYoshihiroFujiwaraDepartment of Orthopaedic Surgery, Okayama Rosai HospitalTakuyaTaokaDepartment of Orthopaedic Surgery, Okayama Rosai HospitalAkiyoshiMiyamotoDepartment of Orthopaedic Surgery, Okayama Rosai HospitalCase Report10.18926/AMO/67878This report presents a new unilateral biportal endoscopic (UBE) technique for lumbar disc herniation without C-arm guidance. Lumbar disc herniation requires surgical intervention when conservative methods fail. Shifts towards minimally invasive percutaneous endoscopic lumbar discectomy, including uniportal and biportal approaches, have been hindered by challenges such as steep learning curves and reliance on radiation-intensive C-arm guidance. We here describe the use of standard intraoperative navigation in UBE to reduce radiation exposure and increase surgical accuracy. A 24-year-old man with low back and bilateral leg pain with gait disturbance was referred to our hospital. He had had conservative treatment for 12 months in another hospital before admission, but this proved unsuccessful. On admission he had low back pain (VAS 4/10) and bilateral leg pain (VAS 8/10), muscle weakness of the bilateral legs (manual muscle testing (MMT) grade of the extensor hallucis longus: 4/4), and numbness of the bilateral lower legs. Preoperative lumbar MRI showed L4/5 large central disc herniation. He underwent C-arm free UBE discectomy under the guidance of O-arm navigation. The surgery was successful, with postoperative lumbar MRI showing good decompression of the dural sac and bilateral L5 nerve roots. The MMT grade and sensory function of both legs had recovered fully on final follow-up at one year. The new UBE technique under navigation guidance was shown to be useful for lumbar disc herniation. This innovative technique was safe and accurate for the treatment of lumbar intervertebral disc herniation, and minimized radiation exposure to surgeons.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7862024Treatment of Tenosynovial Giant Cell Tumor of the Cervical Spine with Postoperative Anti-RANKL Antibody (Denosumab) Administration469474ENYuichiHirataDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTakayukiNagaseDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSusumuSasadaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshiyukiAyadaDepartment of Pathology and Oncology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHayatoMiyakeDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesChiakiSugaharaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHidetakaYamamotoDepartment of Pathology and Oncology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshinaoOdaDepartment of Anatomic Pathology, Graduate School of Medical Sciences, Kyushu UniversityTakaoYasuharaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShotaTanakaDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/67877Tenosynovial giant cell tumor (TGCT) is a fibrous histiocytic tumor originating in the synovial membrane. While cervical TGCT may not be considered a common diagnosis preoperatively because it is relatively rare, it has a high recurrence rate and should be considered. Total resection is preferable, but it can be challenging due to the risk of damaging the vertebral artery. Denosumab has shown effectiveness as a postoperative treatment for osteolytic bone lesion. Denosumab administration coupled with close follow-up might offer an effective postoperative treatment option for unresectable TGCT with bone invasion.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7862024Secondary Polymyalgia Rheumatica Following SARS-CoV-2 Infection465468ENKazukiOchoDepartment of Internal Medicine, Ishikawa HospitalHideharuHagiyaDepartment of Infectious Diseases, Okayama University HospitalHisashiIshikawaDepartment of Internal Medicine, Ishikawa HospitalFumioOtsukaDepartment of General Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/67873An 81-year-old Japanese man with a medical history of diabetes mellitus and hypertension was diagnosed with the novel coronavirus disease 2019 (COVID-19). The patient developed pain in the bilateral shoulders and hips 3 days after the disease onset and presented to our outpatient clinic after 1 month. Referring to diagnostic criteria, we diagnosed him with polymyalgia rheumatica (PMR). We initiated prednisolone at 15 mg per day and his symptoms improved immediately. The clinical course of the patient indicated that the SARS-CoV-2 infection triggered the onset of autoimmune disease, PMR in this case.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7862024Traumatic Neuroma Arising from Surgical Trauma during Conversion from Laparoscopic to Open Cholecystectomy459464ENShinyaSakamotoDepartment of Gastroenterological Surgery, Kochi Health Sciences CenterMotoyasuTabuchiDepartment of Gastroenterological Surgery, Kochi Health Sciences CenterRikaYoshimatsuDepartment of Radiology, Kochi Health Sciences CenterManabuMatsumotoDepartment of Diagnostic Pathology, Kochi Health Sciences CenterJunIwataDepartment of Diagnostic Pathology, Kochi Health Sciences CenterTakehiroOkabayashiDepartment of Gastroenterological Surgery, Kochi Health Sciences CenterCase Report10.18926/AMO/67872Traumatic neuroma is an abnormal proliferation of injured nerves resulting from trauma or surgery. We present a case of traumatic neuroma arising in the cystic duct after cholecystectomy. A 66-year-old man was referred to our department due to a biliary tumor. He had undergone cholecystectomy 20 years prior. Cholangioscopy showed an elevated lesion covered with smooth mucosa. Histological examination revealed normal bile duct mucosa. Although benign disease was suspected, the possibilities of malignant disease could not be excluded. Extrahepatic bile duct resection was planned to include intraoperative rapid-freezing of a biopsy specimen followed by histopathological examination. These intraoperative histology results showed proliferation of nerve and fibrous tissue only, resulting in the diagnosis of traumatic neuroma, so no lymph nodes were removed. To avoid excessive surgical intervention, histopathological examination of an intraoperative rapid-frozen biopsy specimen may be important for diagnosing traumatic neuroma.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7862024Case of Radiation-Induced Angiosarcoma after Breast-Conserving Surgery with Hypofractionated Radiotherapy in a Japanese Patient453458ENYujiroKawataDepartment of Radiology, Kawasaki Medical SchoolKentaWatanabeDepartment of Radiology, Kawasaki Medical SchoolRyojiTokiyaDepartment of Radiology, Kawasaki Medical SchoolTakeshiMatsunoDepartment of Pathology, Kawasaki Medical SchoolRyoTanakaDepartment of Dermatology, Kawasaki Medical SchoolNarutoTairaDepartment of Breast and Thyroid Surgery, Kawasaki Medical SchoolKuniakiKatsuiDepartment of Radiology, Kawasaki Medical SchoolCase Report10.18926/AMO/67871Radiation-induced angiosarcoma (RIAS) is a rare, late adverse event of radiotherapy comprising approximately half of all radiation-induced sarcomas. It has a relatively short latency period and generally unfavorable prognosis. This study presents a case of RIAS that developed 5 years and 11 months after the completion of hypofractionated radiotherapy (42.56 Gy/16 fractions) following partial mastectomy. The patient was diagnosed with RIAS 10 months after the onset of skin redness. She underwent skin tumor resection, followed by paclitaxel, then pazopanib administration, but no radiotherapy. At 6 years and 2 months after surgery, no RIAS recurrence has been detected.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7862024Closure of Ventricular Septal Rupture through a Left Thoracotomy in a Patient with a History of Esophageal Reconstruction449452ENGentaroKatoDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalTatsuyaOgawaDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalTomohiroHayashidaDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalShujiShimizuDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences and Okayama University HospitalShuYamamotoDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalTakeshiShichijoDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalCase Report10.18926/AMO/67870A 73-year-old man who had undergone esophagectomy and retrosternal gastric tube reconstruction for esophageal cancer 8 years prior was transferred to our hospital for the treatment of an acute myocardial infarction. Emergent percutaneous coronary intervention for the left anterior descending artery (#7) was successfully performed. However, echocardiography revealed a ventricular septal rupture (25×27 mm). Seventeen days after admission, the rupture was successfully treated with a double-patch closure via a left anterolateral thoracotomy to avoid a surgical injury to his retrosternal gastric tube. Determining the best surgical approach to the heart is important for safe cardiac surgery in patients after esophageal reconstruction.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2192-31831412024Successful immunotherapy with ipilimumab and nivolumab in a patient with pulmonary sclerosing pneumocytoma6063ENYumiInukai-MotokuraDepartment of Allergy and Respiratory Medicine, Okayama University HospitalKiichiroNinomiyaDepartment of Allergy and Respiratory Medicine, Okayama University HospitalTakahiroBabaDepartment of Allergy and Respiratory Medicine, Okayama University HospitalHirokiOmoriDepartment of Allergy and Respiratory Medicine, Okayama University HospitalTetsuyaTakeguchiDepartment of Allergy and Respiratory Medicine, Okayama University HospitalMariUnoDepartment of Allergy and Respiratory Medicine, Okayama University HospitalYoshiyukiAyadaDepartment of Pathology, Okayama University HospitalTakehiroTanakaDepartment of Pathology and Oncology, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityYoshinobuMaedaDepartment of Hematology, Oncology and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKadoakiOhashiDepartment of Allergy and Respiratory Medicine, Okayama University HospitalPulmonary sclerosing pneumocytoma (PSP) is a rare form of lung cancer that occasionally presents with lymph node and extrapulmonary metastases, and multiple lesions. The treatment of metastatic PSP remains undefined. This study reports the case of a 48-year-old female patient diagnosed with PSP following surgical intervention for a solitary nodule in the left lower lobe. Four years later, recurrence occurred in the left hilar and mediastinal lymph nodes, necessitating an additional resection. Concurrently, sacral metastases developed and required palliative radiotherapy. Genetic analysis identified an AKT1 E17K mutation, characteristic of PSP, and absence of programmed cell death ligand 1 (PD-L1) expression in the tumor. Two years post-recurrence, the tumor recurred in the left mammary gland and mediastinal lymph nodes. Combination immunotherapy with ipilimumab and nivolumab yielded a significantly positive response in this metastatic PSP case. This is the first reported case of successful treatment of multiple distant metastatic PSP with ipilimumab and nivolumab, following the failure of various local treatments. Further case series are warranted to validate the efficacy of immunotherapy in metastatic PSP.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama2168-818416112024Infective Endocarditis With Origin in Orbital Vascular Malformation and Maxillary Sinusitis: A Case Report and Review of Four Patients in the Literaturee74873ENToshihikoMatsuoDepartment of Ophthalmology, Graduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityYoshitakaIwamotoDepartment of General Internal Medicine, Okayama Medical Center, National Hospital OrganizationHironoriOkamotoDepartment of General Internal Medicine, Okayama Medical Center, National Hospital OrganizationDaisukeIguchiDepartment of Internal Medicine, Ochiai HospitalInfective endocarditis is a life-threatening disease and the early diagnosis is crucial for a better outcome. We report an old adult who developed infective endocarditis in association with new-onset maxillary sinusitis as well as proptosis, which was caused by an orbital mass lesion in the background of pre-existing orbital vascular malformation. A 74-year-old woman was found incidentally to have right orbital vascular (venous) malformation by head magnetic resonance imaging when she was hospitalized for left dorsal pontine infarction. No paranasal sinusitis was noted at that time. She was well until half a year later when she developed fatigue and appetite loss for two days. At the same time, she had proptosis on the right side but did not have a fever. Blood examinations showed leukocytosis and a marked increase of C-reactive protein to 22 mg/dL as well as a moderate increase of bilirubin and liver enzymes. Emergency computed tomography scans from the head to abdomen showed nothing to be noted except for maxillary sinusitis and a retrobulbar orbital mass on the right side, which was in the same location as pre-existing vascular malformation. She began to have empirical antibiotics suspected of infective endocarditis. Head magnetic resonance imaging showed ischemic lesions in the right parietal lobe. Transthoracic and transesophageal echocardiography showed mitral valve regurgitation but no apparent vegetation. Streptococcus anginosus was detected by blood culture and the antibiotics were switched to intravenous penicillin G for 32 days. She was discharged in healthy condition with no proptosis. The orbital vascular malformation might serve as a route for infective endocarditis with the infectious origin in maxillary sinusitis. Maxillary sinusitis would be a predisposing factor for the development of infective endocarditis, and proptosis caused by an infectious focus of abnormal vascular channels in the orbit would lead to the early diagnosis of infective endocarditis. The present patient is unique in showing infective endocarditis in association with orbital vascular malformation.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2090-644720242024A Rare Case of Multiple Myeloma Identified Following the Diagnosis of Amyloidosis of the Tongue8836103ENHidekaKanemotoDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKyoichiObataDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKoichiKadoyaDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKishoOnoDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHotakaKawaiDepartment of Oral Pathology and Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukiKunisadaDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMayumiYaoDepartment of Oral and Maxillofacial Surgery, Tsuyama Chuo HospitalSoichiroIbaragiDepartment of Oral and Maxillofacial Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAmyloidosis is a disease in which amyloid protein is deposited in organs and tissues, resulting in functional impairment. Amyloidosis occurs in 12%-30% of patients with multiple myeloma, but in rare cases, amyloidosis may precede the diagnosis of multiple myeloma. Our patient was a 76-year-old Japanese male on dialysis. Multiple nodules accompanied by ulcers were observed on his tongue. He had no subjective symptoms or clinical findings associated with multiple myeloma. The histopathological findings suggested amyloidosis. We suspected both systemic and localized amyloidosis and performed a comprehensive systemic examination. Since the patient had been on dialysis for only a short period of time (similar to 3 months), dialysis-related amyloidosis was ruled out. After blood and urine tests, a diagnosis of multiple myeloma was made. Chemotherapy treatment was started, but the patient's multiple myeloma could not be suppressed and the tongue amyloidosis worsened, leading to his death 2 years and 2 months after the initial diagnosis.No potential conflict of interest relevant to this article was reported.Thieme GruppeActa Medica Okayama2364-372212112024Analysis of painful situations during unsedated esophagogastroduodenoscopyE1267E1276ENHiromitsuKanzakiGastroenterology and Hepatology, Okayama University HospitalSakikoKuraokaGastroenterology and Hepatology, Okayama University HospitalTakuyaSatomiGastroenterology and Hepatology, Okayama University HospitalShotaroOkanoueGastroenterology and Hepatology, Okayama University HospitalKentaHamadaPractical Gastrointestinal Endoscopy, Okayama UniversityYoshiyasuKonoGastroenterology and Hepatology, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesMasayaIwamuroGastroenterology and Hepatology, Okayama University HospitalSeijiKawanoGastroenterology and Hepatology, Okayama University HospitalYoshiroKawaharaPractical Gastrointestinal Endoscopy, Okayama UniversityHiroyukiOkadaGastroenterology and Hepatology, Okayama University HospitalMotoyukiOtsukaGastroenterology and Hepatology, Okayama University Graduate School of Medicine Dentistry and Pharmaceutical SciencesBackground and study aims Although esophagogastroduodenoscopy (EGD) is a widely used technique, the procedure is often associated with discomfort. This study aimed to analyze painful situations, their frequency, and factors associated with patient discomfort during EGD. <br>
Patients and methods This prospective observational study included patients scheduled to undergo EGD. Seven endoscopists recruited patients scheduled for EGD screening or surveillance. Each endoscopist enrolled 20 patients, performing 10 EGD procedures using ultraslim endoscopes and 10 with standard-sized endoscopes. Data regarding painful situations and frequency were collected using specialized buttons pressed by the patients during EGD. A survey about overall discomfort was conducted after the procedure. <br>
Results We analyzed data from 140 patients. Esophageal insertion and duodenal observation were associated with the highest incidence of pressing the pain button, accounting for 59.3% and 40.7% of the cases, respectively. The factor associated with pressing the pain button during esophageal insertion was endoscopist experience (< 10 years). In contrast, younger age and female sex were the factors associated with pressing the pain button during duodenal observation. In the post-procedure survey, 63.6% of patients reported discomfort. Factors associated with patient discomfort included pressing the pain button during esophageal insertion (odds ratio [OR]: 2.84, P = 0.01) and previous painful EGD experience (OR: 2.41, P = 0.03). <br>
Concusions This study provides objective data on painful situations, their frequency, and related factors during EGD. Further research and interventions focusing on pain reduction during endoscopic procedures are warranted. The results of this study will help endoscopists manage painful situations and potentially improve skills.No potential conflict of interest relevant to this article was reported.Japanese Society of Internal MedicineActa Medica Okayama0918-291863192024A Prompt Diagnosis and Treatment of a Case of Nuclear Protein of the Testis Carcinoma Characterized by a Bronchial Lesion and High Serum Alpha-fetoprotein Level Following Genomic Testing26552660ENHiroakiMatsuuraDepartment of Respiratory Medicine, Okayama University HospitalGoMakimotoDepartment of Respiratory Medicine, Okayama University HospitalNaohiroOdaDepartment of Respiratory Medicine, Fukuyama City HospitalKiichiroNinomiyaCenter for Comprehensive Genomic Medicine, Okayama University HospitalHisaoHigoDepartment of Respiratory Medicine, Okayama University HospitalMasanoriFujiiDepartment of Respiratory Medicine, Okayama University HospitalKammeiRaiCenter for Innovative Clinical Medicine, Okayama University HospitalEikiIchiharaDepartment of Respiratory Medicine, Okayama University HospitalKadoakiOhashiDepartment of Respiratory Medicine, Okayama University HospitalKatsuyukiHottaCenter for Innovative Clinical Medicine, Okayama University HospitalMasahiroTabataCenter for Clinical Oncology, Okayama University HospitalYoshinobuMaedaDepartment of Hematology, Oncology, and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesNuclear protein of the testis carcinoma (NUTC) is a rare and aggressive malignancy. We herein report a case of NUTC in the lung characterized by a bronchial lesion and elevated alpha-fetoprotein levels. A 35-year-old Japanese man presented to our institution with suspected advanced lung cancer based on a histological examination. Subsequently, next-generation sequencing (NGS) yielded a positive BRD4-NUTM1 fusion. In addition, positive NUT immunostaining of the lung biopsy specimen confirmed NUTC in the lungs. Systemic chemotherapy and radiotherapy showed a temporary response, with decreased serum alpha-fetoprotein levels. We highlight this case of a prompt diagnosis by NGS of NUTC in a young individual with a rapidly progressing tumor.No potential conflict of interest relevant to this article was reported.岡山大学経済学会Acta Medica Okayama2433-41465622024学生による地方創生活動10年の成果と課題―瀬戸内市裳掛地区での実践活動―1740ENSatoshiMimuraAkaneTokoo10.18926/OER/67706 岡山大学の公認学生サークルのなかで地方創生活動を展開しているサークルに「まちづくり研究会」がある。岡山大学学生サークル「まちづくり研究会」は，2014年に活動を開始，その活動の目的は，過疎化人口減少の問題を抱える瀬戸内市邑久町虫明（おくちょうむしあけ）の裳掛（もかけ）地区をフィールドとした学生と地域住民による地方創生の協働活動であり，通称『瀬戸内市裳掛地区再生プロジェクト』と呼ばれている。
この10年間，学生たちは先輩たちから後輩たちに実践活動をリレーしながら様々な活動を展開してきた。その主眼が，生徒数の減る裳掛小学校の子どもたちの支援活動を続けることで，生徒数の減少に歯止めをかけることである。そのための具体的な方策として，「学生による情報発信」，「耕作放棄地対策への参加」，「空き家の再生支援による拠点づくり」，「移住定住促進支援」，「地域催事への企画・運営支援」，「子どもたちの見守り」などの活動を実践しながら地域を元気にする応援を継続してきた。なお，掲載写真の使用許可は，活動の都度，地域の皆様（保護者を含む）や学生たちに確認・了解を得ている。
本論では，この学生たちの活動の成果を紹介しながら，学生たちの学びと地域への貢献という互恵性の視座に立ち「学生による地域創生活動の実践事例研究」として報告する。No potential conflict of interest relevant to this article was reported.SpringerActa Medica Okayama2198-77931012024Gastro-tracheal fistula following esophageal cancer surgery through the retrosternal route: a case report252ENSeitaroNishimuraDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKazuhiroNomaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKentoKawasakiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineMasashiHashimotoDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineTakuyaKatoDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineNaoakiMaedaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShunsukeTanabeDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYasuhiroShirakawaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineBackground Gastro-tracheal fistula is a rare but serious complication after esophageal surgery, often requiring long-term treatment and invasive procedures. Gastro-tracheal fistula usually occurs through the posterior mediastinal route and rarely through the retrosternal route. No previous reports have described gastro-tracheal fistula after retrosternal route reconstruction was cured by conservative treatment.<br>
Case presentation A 70-year-old man with lower thoracic esophageal cancer underwent thoracoscopic esophagectomy in the prone position and gastric tube reconstruction through the retrosternal route with neck anastomosis after neoadjuvant chemotherapy. Despite anastomotic leakage on postoperative day 10, his general condition was stable, and he was managed conservatively with antibiotics and gastric tube decompression. On day 29, he presented with high fever and a gastro-tracheal fistula was observed by esophagography. Conservative management was continued because the patient remained stable. On day 48, esophagography showed that the fistula was undetectable. The patient was able to take fluids orally. He progressed well on an oral diet and was transferred to a different hospital.<br>
Conclusions A gastro-tracheal fistula, although rare, can occur after retrosternal route reconstruction. When a patient is stable, gastro-tracheal fistula after retrosternal route reconstruction may be cured by conservative treatment.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama2352-409X602024Petrological characterization for material provenance of haniwa earthenware from mounded tombs in the Kibi region, Japan104813ENToshioNozakaDepartment of Earth Sciences, Okayama UniversityNaoyaOhbayashiDepartment of Earth Sciences, Okayama UniversityYukiTodaDepartment of Earth Sciences, Okayama UniversityKanakoSugiuraDepartment of Archaeology, Okayama UniversityTakahiroNozakiResearch Institute for the Dynamics of Civilizations, Okayama UniversityOsamuKimuraResearch Institute for the Dynamics of Civilizations, Okayama UniversityNaokoMatsumotoResearch Institute for the Dynamics of Civilizations, Okayama UniversityAkiraSeikeDepartment of Archaeology, Okayama UniversityTo determine the provenance of the materials used in the production of haniwa earthenware unearthed from mounded tombs (kofun) in the Kibi region (modern Okayama Prefecture) during the Kofun period (late 3rd – 6th century CE) of Japan, we carried out petrological analyses of haniwa sherds, including optical microscopy, X-ray diffractometry, X-ray fluorescence spectroscopy, and electron-probe analysis. The 25 haniwa sherds analyzed from 12 representative mounded tombs are composed of mineral and rock inclusions with variable grain size set in a clay matrix. The dominant inclusions are quartz, K-feldspar, and plagioclase, associated with minor amounts of amphibole, volcanic glass, and granitic rocks in all the haniwa sherds, and small amounts of hornfels, quartz rock, and accessory minerals, including mica, ilmenite, and chromite, in some of the sherds. Amphibole and plagioclase have compositional variations indicative of the mixing of tephra and granitic components. The compositions of volcanic glass inclusions are similar to those of the Aira-Tanzawa and Kikai-Akahoya tephras widely distributed in southwestern Japan. Bulk chemical compositions show magmatic differentiation trends, which are variable between individual tombs. From these results, it is concluded that the paste materials of haniwa in the Kibi region were commonly derived from weathered granitic rocks mixed with minor amounts of three widespread tephras. The variations of chemical and mineralogical compositions are probably the reflection of local geologic settings, suggesting the presence of specific mining sites of paste materials around each tomb. The mining sites could be located at the bases of hills of granitic rocks covered by widespread tephras and in some cases, near the flood plain of big river systems.No potential conflict of interest relevant to this article was reported.Nature PortfolioActa Medica Okayama2045-23221412024Perioperative gum-chewing training prevents a decrease in tongue pressure after esophagectomy in thoracic esophageal cancer patients: a nonrandomized trial23886ENReikoYamanaka-KohnoDepartment of Preventive Dentistry, Division of Dentistry, Okayama University HospitalYasuhiroShirakawaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical ScienceAyaYokoiDepartment of Preventive Dentistry, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityNaoakiMaedaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical ScienceShunsukeTanabeDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical ScienceKazuhiroNomaDepartment of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical ScienceKazuyoshiShimizuDepartment of Anesthesiology and Resuscitology, Okayama University HospitalToshiharuMituhashiCenter for Innovative Clinical Medicine, Okayama University HospitalYoshihideNakamuraDepartment of Oral and Maxillofacial Radiology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical ScienceSoutoNanbaDental Clinic, Kurashiki Medical Check-Up CenterYurikaUchidaDepartment of Preventive Dentistry, Division of Dentistry, Okayama University HospitalTakayukiMaruyamaDepartment of Preventive Dentistry, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityManabuMoritaDepartment of Oral Health Sciences, Takarazuka University of Medical and Health CareDaisukeEkuniDepartment of Preventive Dentistry, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTongue pressure (TP) decreases significantly after esophagectomy in esophageal cancer patients (ECPs). Meanwhile, 2 weeks of gum-chewing training (GCT) significantly increased TP in healthy university students. We examined whether perioperative GCT would decrease the proportion of patients exhibiting a decline in TP at 2 weeks postoperatively, and prevent postoperative complications, in thoracic ECPs (TECPs). This was a single-center interventional study, and nonrandomized study with a historical control group (HCG). TECPs who underwent first-stage radical esophagectomy were recruited. Thirty-two patients of 40 in the gum-chewing group (GCG) were completed perioperative GCT in 3 times daily. Propensity score matching was performed with covariates related to TP including preoperative age, sex, body mass index, and the repetitive saliva swallowing test result, and yielded a matched cohort of 25 case pairs. Eleven GCG patients [44.0%] exhibited significantly lower TP at 2 weeks postoperatively than before esophagectomy was significantly fewer than that of 19 patients [76.0%] in the HCG. The median number of fever days (> 38 degrees C) in the 2 weeks after esophagectomy in the GCG was significantly fewer than those in the HCG. Perioperative GCT may prevent postoperative TP decline and postoperative dysphagia-related complications after esophagectomy.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7852024Occult Nesidioblastosis Detected by 111In-Pentetreotide Single-Photon Emission Computed Tomography423428ENShinyaSakamotoDepartment of Gastroenteorlogical Surgery, Kochi Health Sciences CenterMotoyasuTabuchiDepartment of Gastroenteorlogical Surgery, Kochi Health Sciences CenterRikaYoshimatsuDepartment of Radiology, Kochi Health Sciences CenterAiHishidaDepartment of Endocrinology and Metabolism, Kochi Health Sciences CenterManabuMatsumotoDepartment of Diagnostic Pathology, Kochi Health Sciences CenterJunIwataDepartment of Diagnostic Pathology, Kochi Health Sciences CenterTakehiroOkabayashiDepartment of Gastroenteorlogical Surgery, Kochi Health Sciences CenterCase Report10.18926/AMO/67667Nesidioblastosis, also known as persistent hyperinsulinemic hypoglycemia, is usually observed in children and infants, although more recently adult-onset nesidioblastosis has also been described. We present a case of nesidioblastosis in a 78-year-old man that was detected by 111In-pentetreotide single photon emission computed tomography (SPECT/CT). The patient was transferred to our hospital’s emergency department in a hypoglycemic coma. Dynamic enhanced CT could detect no lesion in the pancreas, but an 111In-pentetreotide SPECT/CT scan performed after a similar episode four weeks later showed increased focal uptake at the head of the pancreas. The results of a selective arterial calcium injection test were negative. After careful consideration and discussion among colleagues, surgical intervention was selected, and a pancreaticoduodenectomy was performed. On histology, there were elevated numbers of Langerhans islets in the pancreatic head, and the islets themselves appeared enlarged. Hypertrophic β-cells comprised the majority, but α-cells, δ-cells and pancreatic polypeptide were also detected in the islets. Based on the histopathological results and repeated hyperinsulinemic hypoglycemic crises, the patient was finally diagnosed with adult-onset nesidioblastosis. He had no hypoglycemic symptoms during outpatient follow-up examination. Since 111In-pentetreotide SPECT/CT may be able to detect nesidioblastosis, clinicians should consider this relatively new-modality examination when encountering such cases.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7852024Focal Cerebral Hypoperfusion Detected by Arterial Spin-Labeled Magnetic Resonance Imaging in Patients with Migraine Presenting with Neurological Symptoms Concomitant with or without Headache413421ENKenichiKashiharaOkayama Neurology ClinicMinoruIrisawaDepartment of Radiology, Okayama Kyokuto HospitalWataruTakaoDivision of Radiology, Okayama Kyokuto HospitalCase Report10.18926/AMO/67666We investigated patients with migraine or migraine variants who exhibited focal cerebral hypoperfusion on arterial spin-labeled (ASL) magnetic resonance (MR) imaging along with neurological symptoms. Fourteen patients with migraine demonstrated focal cerebral hypoperfusion. Three other patients did not have a history of recurrent headaches but exhibited comparable cerebral hypoperfusion to migraine patients on ASL-MRI in addition to neurological symptoms. Patients with migraine may present with neurological symptoms associated with cortical spreading depression during, after, or even without a headache phase. Additionally, the isolated neurological symptoms may be caused by a pathophysiology identical to that of migraine but without presenting with recurrent headaches.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7852024The First Report of Bickerstaff Brainstem Encephalitis Induced by Atezolizumab for Metastatic Breast Cancer407412ENKyokoShimoyamaDepartment of Breast Surgery, Takatsuki General HospitalAtsushiNakajimaDepartment of Rehabilitation, Aijinkai Rehabilitation HospitalYoshimitsuMinariDepartment of Breast Surgery, Takatsuki General HospitalCase Report10.18926/AMO/67665Immune checkpoint inhibitors (ICIs) have revolutionized cancer treatment, but they have been known to cause immune-related adverse events (irAEs) by promoting T-cell activation. Neurological irAEs are rare (1%) but have a high fatality rate (11.5%). Here we report the first case of Bickerstaff brainstem encephalitis (BBE) induced by an ICI. A woman in her 60s with metastatic breast cancer was treated with atezolizumab plus nab-paclitaxel once intravenously. Eighteen days later, she lost consciousness with ophthalmoplegia and was diagnosed with a neurological irAE. She recovered consciousness immediately with the administration of intravenous immunoglobulin (IVIG) but suffered severe permanent peripheral neuropathy. Although it is just one case, this experience shows that BBE occurring as a neurological irAE of ICI cancer treatment may be associated with more severe outcomes than conventional BBE in metastatic cancer. Creating a system for multidisciplinary treatment is essential for ICI therapy.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7852024Pediatric Severe Febrile Thrombocytopenia Syndrome: A Case Report and Literature Review401405ENYusukeToyotaDepartment of Pediatrics, NHO Iwakuni Clinical CenterKazuhiroUdaDepartment of Pediatrics, NHO Iwakuni Clinical CenterKomeiShirabeYamaguchi Prefectural Institute of Public Health and EnvironmentTadashiMoriwakeDepartment of Pediatrics, NHO Iwakuni Clinical CenterCase Report10.18926/AMO/67664Severe febrile thrombocytopenia syndrome (SFTS) is a tick-borne infectious disease that is endemic in parts of eastern Asia. Few pediatric cases have been reported. We describe a case of SFTS in a seven-year-old girl who presented with prolonged fever and gastrointestinal symptoms. Leukopenia and thrombocytopenia on hematology, and a history of outdoor activity led us to diagnose SFTS, although the patient had no tick bite marks. We also review the literature and discuss the characteristics of pediatric SFTS. Physicians should consider SFTS in the differential diagnosis of fever with thrombocytopenia in children living in endemic areas.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama0815-931939122024Rates and risk factors of bleeding after gastric endoscopic submucosal dissection with continuous warfarin or 1‐day withdrawal of direct oral anticoagulants27602766ENShoichiroHirataDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKentaHamadaDepartment of Practical Gastrointestinal Endoscopy, Okayama UniversityMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHirokazuMouriDepartment of Gastroenterology and Hepatology, Kurashiki Central HospitalKojiMiyaharaDepartment of Internal Medicine, Hiroshima City HospitalTakaoTsuzukiDepartment of Gastroenterology, Japanese Red Cross Society Himeji HospitalKenjiYamauchiDepartment of Gastroenterology, Mitoyo General HospitalSayoKobayashiDepartment of Internal Medicine, Fukuyama City HospitalSakumaTakahashiDepartment of Gastroenterology, Kagawa Prefectural Central HospitalRyutaTakenakaDepartment of Internal Medicine, Tsuyama Chuo HospitalShinichiroHoriDepartment of Gastroenterology, Japanese Red Cross Society Himeji HospitalMasafumiInoueDepartment of Gastroenterology, Japanese Red Cross Okayama HospitalTatsuyaToyokawaDepartment of Gastroenterology, National Hospital Organization Fukuyama Medical CenterMamoruNishimuraDepartment of Internal Medicine, Okayama City HospitalShuheiIshiyamaDepartment of Internal Medicine, Okayama Saiseikai General HospitalJiroMiyaikeDepartment of Internal Medicine, Saiseikai Imabari HospitalRyoKatoDepartment of Gastroenterology, National Hospital Organization Iwakuni Clinical CenterMinoruMatsubaraDepartment of Internal Medicine, Sumitomo Besshi HospitalNaokoYunokiDepartment of Internal Medicine, Akaiwa Medical Association HospitalHiromitsuKanzakiDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshiroKawaharaDepartment of Practical Gastrointestinal Endoscopy, Okayama UniversityHiroyukiOkadaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHidekiIshikawaDepartment of Molecular-Targeting Prevention, Kyoto Prefectural University of MedicineMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesOkayama Gut Study GroupBackground and Aim: The 2017 Japanese guidelines recommend continuing warfarin therapy during the perioperative period or discontinuing direct oral anticoagulants (DOACs) only on the day of endoscopic submucosal dissection for early gastric cancer. However, their safety has not been sufficiently explored. This study aimed to validate this management method.<br>
Methods: This retrospective, multicenter study analyzed the characteristics and outcomes of patients who underwent gastric endoscopic submucosal dissection between July 2017 and June 2019. The patients were categorized according to the use of warfarin or DOACs.<br>
Results: Among the 62 eligible patients, 53 (85%) were male (median age, 76 years). Warfarin was used in 10 patients (16%) and DOACs in 52 patients (84%). Fourteen patients taking DOACs (27%) used concomitant antiplatelet agents, with seven patients (13%) continuing treatment at the time of the endoscopic procedure. No postprocedural bleeding occurred in patients receiving warfarin (0%), whereas 10 cases (19%) of bleeding occurred in patients receiving DOACs: rivaroxaban, 0% (0/22); dabigatran, 0% (0/2); edoxaban, 43% (6/14); and apixaban, 29% (4/14). The type of anticoagulant (P < 0.01) and continuation of antiplatelet therapy (P = 0.02) were risk factors for postprocedural bleeding in patients receiving DOACs. Intraprocedural bleeding requiring transfusion or symptomatic thromboembolic events were not reported.<br>
Conclusions: Continuous warfarin therapy is preferred. DOAC withdrawal 1 day before a procedure is associated with a high bleeding rate, which may differ for different types of anticoagulants. The continuation of antiplatelet medications in patients receiving DOACs carries a high risk of bleeding and is a future challenge.No potential conflict of interest relevant to this article was reported.American Society of Clinical Oncology (ASCO)Acta Medica Okayama2473-428482024Response to Imatinib in a Patient With Gastric Adenocarcinoma With KIT Q556_K558 In-Frame Deletion: A Case Reporte2400228ENKiichiroNinomiyaCenter for Comprehensive Genomic Medicine, Okayama University HospitalDaisukeEnnishiCenter for Comprehensive Genomic Medicine, Okayama University HospitalKunioOkamotoDepartment of Medical Oncology, Kagawa Prefectural Central HospitalMidoriAndoDepartment of Pathology, Kagawa Prefectural Central Hospital,SatokoNakamuraDepartment of Pathology, Kagawa Prefectural Central HospitalShutaTomidaCenter for Comprehensive Genomic Medicine, Okayama University HospitalYoshiyukiAyadaDepartment of Pathology, Okayama University HospitalGoMakimotoCenter for Clinical Oncology, Okayama University HospitalEikiIchiharaCenter for Clinical Oncology, Okayama University HospitalNatsukoOkitaResearch Management Division, Clinical Research Support Office, National Cancer Center HospitalShinichiToyookaCenter for Comprehensive Genomic Medicine, Okayama University HospitalYoshinobuMaedaDepartment of Hematology, Oncology and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMasahiroTabataCenter for Clinical Oncology, Okayama University HospitalNo potential conflict of interest relevant to this article was reported.SpringerActa Medica Okayama2198-77931012024Primary ileal myeloid sarcoma presenting with bowel obstruction: a case report229ENHitoshiMinagiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineNobuhikoKanayaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYoshitakaKondoDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYoshihikoKakiuchiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShinjiKurodaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineRyoheiShojiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineHajimeKashimaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineYukiMatsumiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineSatoruKikuchiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineKunitoshiShigeyasuDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineFuminoriTeraishiDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineShunsukeKagawaDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineToshiyoshiFujiwaraDepartment of Gastroenterological Surgery, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineBackground Myeloid sarcoma (MS) is an extramedullary tumor constituted by myeloid blasts or immature myeloid cells. It frequently occurs in conjunction with acute myeloid leukemia (AML); however, it can exceptionally manifest in patients without leukemia. Here, we present a rare case of primary MS originating in the small bowel without evidence of bone marrow involvement.<br>
Case representation A 33 year-old female with no relevant medical history was admitted to our hospital with recurrent abdominal pain. Computed tomography (CT) revealed bowel obstruction due to thickening of the ileum wall, which was suspected to be an ileal tumor. Initially, ectopic endometriosis was suspected because of abdominal pain associated with the menstrual cycle and changes observed on a follow-up CT scan. The lesion could not be detected by double-balloon endoscopy. Despite conservative treatment, the obstruction persisted, and laparoscopic partial ileal resection was performed, which revealed extensive involvement of the ileum and mesentery. Additionally, the mesentery of the resected ileum was extremely thickened. Histopathological and immunohistochemical examinations of the surgical specimen indicated ileal MS. Bone marrow aspiration after discharge was negative for cytological findings of leukemia, leading to a final diagnosis of primary ileal MS. Her postoperative course was uneventful, and she is currently undergoing systemic chemotherapy tailored to AML at another hospital.<br>
Conclusions Even though MS of the small bowel is rare and may not be considered preoperatively, similar surgical treatment to that of other small bowel malignancies can ensure proper postoperative diagnosis and appropriate chemotherapy. Given the potential need for chemotherapy, ensuring surgical safety that allows for its rapid initiation is critical.No potential conflict of interest relevant to this article was reported.Elmer Press, Inc.Acta Medica Okayama1923-415515112024Intravitreal Fluconazole Injection for Fungal Endophthalmitis as Treatment Option in a Patient With End-Stage Liver and Kidney Diseases359366ENToshihikoMatsuoGraduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityYasuyukiKobayashiDepartment of Urology, Okayama University HospitalShingoNishimuraDepartment of Urology, Okayama University HospitalNaokoYoshiokaDepartment of Gastroenterology and Hepatology, Kawasaki Medical SchoolYasushiTakahashiDepartment of Internal Medicine, Ochiai HospitalYasutakaIguchiDepartment of Internal Medicine, Ochiai HospitalEndogenous endophthalmitis is an infectious disease of the intraocular tissue that is a consequence of bloodstream infection. The efficacy of intravitreal fluconazole injection to assist low-dose oral fluconazole in fungal endophthalmitis remains unknown in older adults with advanced liver and renal disease. In this case report, a 78-year-old man with hepatitis C virus-related liver cirrhosis and hepatocellular carcinoma who also had end-stage renal disease with temporary nephrostomy noticed blurred vision and showed a large retinal infiltrate with vitreous opacity in the right eye. In the clinical diagnosis of endogenous fungal endophthalmitis, he had an intravitreal injection of 0.1% fluconazole in 0.2 - 0.3 mL every 2 weeks four times in total, in addition to a minimum dose of oral fluconazole. One month before the ophthalmic presentation, he developed a fever and computed tomography scan showed ureterolithiasis with hydronephrosis on the right side, indicating that the renal pelvic stone fell into the ureter. He underwent nephrostomy tube insertion on the right side in the diagnosis of obstructive urinary tract infection. In the course, a potassium hydroxide (KOH) preparation of the urine sediments which were obtained from the nephrostomy tube showed yeast-like fungi, suggestive of Candida, 1 week before the development of eye symptoms. One week after the ophthalmic presentation, the nephrostomy tube at 14 Fr (French gauge) which had been inserted 1 month previously was replaced with a new tube with a larger size at 16 Fr because urine excretion from the tube was reduced. Immediately after the exchange of the nephrostomy tube, a large volume of urine was excreted from the tube. In a week, he had no systemic symptoms and serum C-reactive protein became low. In the meantime, the retinal infiltrate became inactive and vitreous opacity resolved. Intravitreal fluconazole injection is a treatment option for fungal endophthalmitis in the case that a patient cannot undergo vitrectomy and cannot take a maximum dose of fluconazole because of poor renal function.No potential conflict of interest relevant to this article was reported.Elsevier BVActa Medica Okayama0385-81465152024Endoscopic Transpterygoid Repair of Sphenoid Sinus Meningocele: A Comprehensive Case Report and Literature Review875879ENAikoShimizuDepartment of Otolaryngology-Head & Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSeiichiroMakiharaDepartment of Otolaryngology-Head & Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyojiImotoDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKojiHirashitaDepartment of Neurosurgery, Kagawa Rosai HospitalMizuoAndoDepartment of Otolaryngology-Head & Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesWe report a challenging and uncommon case involving a 53-year-old Japanese man with cerebrospinal fluid (CSF) leakage caused by a meningocele in the lateral recess of the sphenoid sinus. Our innovative treatment approach involved a combination of transpterygoid and endoscopic modified medial maxillectomy techniques, with special emphasis on the preservation of the sphenopalatine artery. This strategic preservation was pivotal to the successful use of the ipsilateral nasoseptal flap for reconstruction, which played a crucial role in the prevention of postoperative CSF leakage. Otolaryngologists and neurosurgeons collaborated to perform the bath-plugging technique; effective collaboration was instrumental to the success of the procedure. This report highlights significant advancement from conventional frontal craniotomy to a more sophisticated endoscopic technique, shows the importance of meticulous surgical planning and execution, emphasizes careful preservation of critical anatomical structures during complex neurosurgical and otolaryngological procedures, and underscores the evolving landscape of surgical approaches for managing complex medical conditions.No potential conflict of interest relevant to this article was reported.SpringerActa Medica Okayama2198-77931012024Central pancreatectomy of the remnant pancreas without reconstruction after pancreatoduodenectomy214ENKinjiHirono Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKoseiTakagi Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesMotohikoYamada Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesJiroKimura Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTomokazuFuji Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKazuyaYasui Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesTakeyoshiNishiyama Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesYasuoNagai Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesNoriyukiKanehira Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesToshiyoshiFujiwara Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesBackground There are several reports on the safety and feasibility of pancreatoduodenectomy (PD) without reconstruction of the small remnant pancreas. However, a few studies have explored central pancreatectomy (CP) for non-reconstructed small remnant pancreases after PD. This study presents a case of CP without pancreatic reconstruction after PD.<br>
Case presentation A 58-year-old man with cerebral palsy underwent PD for distal cholangiocarcinoma. Three years postoperatively, a 12-mm tumor was detected in the remnant pancreatic body and diagnosed as a pancreatic neuroendocrine neoplasm. Surgical resection was performed, because the tumor was enlarged and chemotherapy resistant. The afferent loop with pancreatojejunostomy anastomosis was dissected, and CP, including pancreatojejunostomy anastomosis, was performed. Given the remnant pancreas was hard and atrophic, the pancreatic tail was transected using a stapler without reconstructing the small remnant pancreas. The patient experienced no postoperative complications including postoperative pancreatic fistula, and the endocrine function of the pancreas was preserved.<br>
Conclusions We present a case of remnant pancreatic CP that did not require reconstruction after PD. Preservation of the small remnant pancreas without reconstruction during CP may be feasible to maintain endocrine function in select patients after PD.No potential conflict of interest relevant to this article was reported.ElsevierActa Medica Okayama2213-0071512024Protracted coronavirus disease 2019 after chimeric antigen receptor-T cell therapy successfully treated with sequential multidrug therapy102104ENMasahiroYamashitaDepartment of Allergy and Respiratory Medicine, Okayama University HospitalHisaoHigoDepartment of Allergy and Respiratory Medicine, Okayama University HospitalNobuharuFujiiDepartment of Hematology and Oncology, Okayama University HospitalChiakiMatsumotoDepartment of Allergy and Respiratory Medicine, Okayama University HospitalGoMakimotoDepartment of Allergy and Respiratory Medicine, Okayama University HospitalKiichiroNinomiyaCenter for Comprehensive Genomic Medicine, Okayama University HospitalMasanoriFujiiDepartment of Allergy and Respiratory Medicine, Okayama University HospitalKammeiRaiCenter for Innovative Clinical Medicine, Okayama University HospitalEikiIchiharaCenter for Clinical Oncology, Okayama University HospitalKadoakiOhashiDepartment of Allergy and Respiratory Medicine, Okayama University HospitalKatsuyukiHottaCenter for Innovative Clinical Medicine, Okayama University HospitalMasahiroTabataCenter for Clinical Oncology, Okayama University HospitalYoshinobuMaedaDepartment of Hematology, Oncology and Respiratory Medicine, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesNobuakiMiyaharaDepartment of Allergy and Respiratory Medicine, Okayama University HospitalA 56-year-old woman who received CD19 chimeric antigen receptor-T cell therapy for refractory diffuse large B-cell lymphoma developed severe coronavirus disease 2019 (COVID-19) and was treated with nirmatrelvir/ritonavir in April 2022. However, she experienced persistent fatigue and cough and fever in June. Computed tomography revealed bilateral ground-glass opacities (GGO), and the patient was treated with corticosteroids for organizing pneumonia after COVID19. Partial improvement was observed, but new GGO appeared despite corticosteroid therapy. Genome analysis of severe acute respiratory syndrome coronavirus 2 detected Omicron variant BA.1.1.2, which was prevalent at the time of initial infection. The patient was diagnosed with protracted COVID-19 and was treated with remdesivir, molnupiravir, nirmatrelvir/ritonavir, and tixagevimab/cilgavimab. These treatments appeared to contribute to the improvement of protracted COVID-19.No potential conflict of interest relevant to this article was reported.BMJ Publishing GroupActa Medica Okayama2755-97342712024Potential dopaminergic deficit in patients with geriatric psychiatric disorders as revealed by DAT-SPECT: a cross-sectional study19ENShintaroTakenoshitaDepartment of Neuropsychiatry, Okayama University HospitalSeishiTeradaDepartment of Neuropsychiatry, Okayama University Faculty of Medicine Dentistry and Pharmaceutical SciencesKatsuhideKojimaDepartment of Radiology, Okayama University HospitalNaotoNishikawaDepartment of Neuropsychiatry, Okayama University HospitalTomokoMikiDepartment of Neuropsychiatry, Okayama University HospitalOsamuYokotaDepartment of Neuropsychiatry, Okayama University Faculty of Medicine Dentistry and Pharmaceutical SciencesMasakiFujiwaraDepartment of Neuropsychiatry, Okayama University HospitalManabuTakakiDepartment of Neuropsychiatry, Okayama University Faculty of Medicine Dentistry and Pharmaceutical SciencesBackground It has been reported that patients with geriatric psychiatric disorders include many cases of the prodromal stages of neurodegenerative diseases. Abnormal I-123-2 beta-carbomethoxy-3 beta-(4-iodophenyl)-N-(3-fluoropropyl) nortropane dopamine transporter single-photon emission computed tomography (DAT-SPECT) reveals a nigrostriatal dopaminergic deficit and is considered useful to detect dementia with Lewy bodies and Parkinson's disease as well as progressive supranuclear palsy and corticobasal degeneration. We aimed to determine the proportion of cases that are abnormal on DAT-SPECT in patients with geriatric psychiatric disorders and to identify their clinical profile. <br>
Methods The design is a cross-sectional study. Clinical findings of 61 inpatients aged 60 years or older who underwent DAT-SPECT and had been diagnosed with psychiatric disorders, but not neurodegenerative disease or dementia were analysed. <br>
Results 36 of 61 (59%) had abnormal results on DAT-SPECT. 54 of 61 patients who had DAT-SPECT (89%) had undergone I-123-metaiodobenzylguanidine myocardial scintigraphy (I-123-MIBG scintigraphy); 12 of the 54 patients (22.2%) had abnormal findings on I-123-MIBG scintigraphy. There were no cases that were normal on DAT-SPECT and abnormal on I-123-MIBG scintigraphy. DAT-SPECT abnormalities were more frequent in patients with late-onset (55 years and older) psychiatric disorders (69.0%) and depressive disorder (75.7%), especially late-onset depressive disorder (79.3%). <br>
Conclusion Patients with geriatric psychiatric disorders include many cases showing abnormalities on DAT-SPECT. It is suggested that these cases are at high risk of developing neurodegenerative diseases characterised by a dopaminergic deficit. It is possible that patients with geriatric psychiatric disorders with abnormal findings on DAT-SPECT tend to show abnormalities on DAT-SPECT first rather than on I-123-MIBG scintigraphy.No potential conflict of interest relevant to this article was reported.岡山医学会Acta Medica Okayama0030-155813622024インフリキシマブが著効した免疫関連有害事象大腸炎の１例6973ENMasayaIwamuroDepartment of Gastroenterology and Hepatology, Okayama University HospitalSakikoHiraokaInflammatory Bowel Disease Center, Okayama University HospitalMotoyukiOtsukaDepartment of Gastroenterology and Hepatology, Faculty of Medicine, Dentistry and Pharmaceutical Sciences, Okayama University A 52-year-old Japanese man diagnosed with non-small cell lung cancer initiated chemotherapy with tremelimumab, durvalumab, nanoparticle albumin-bound paclitaxel, and carboplatin. On the fourth day of the first treatment course, he developed a fever, followed by watery diarrhea exceeding 10 episodes per day and bloody stools the next day. Immunotherapy-related adverse event colitis was diagnosed through CT scans and colonoscopy examinations. Despite the ineffectiveness of systemic steroid administration, prompt alleviation of symptoms was achieved through the administration of infliximab. In our case, the patient developed Grade 3 diarrhea, prompting the initiation of intravenous prednisolone at 80mg/day in accordance with guidelines. However, symptom improvement was not attained. In situations where symptoms persist beyond three days despite systemic steroid administration, the consideration of adjunctive infliximab use at a dosage of 5mg/kg becomes necessary.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2769-2558332024Forgetfulness in adult attention-deficit/hyperactivity disorder masks transient epileptic amnesia: a case reporte70003ENTakashiFukaoDepartment of Neuropsychiatry, OkayamaUniversity HospitalMasakiFujiwaraDepartment of Neuropsychiatry, OkayamaUniversity HospitalYutoYamadaDepartment of Neuropsychiatry, OkayamaUniversity HospitalShinjiSakamotoDepartment of Neuropsychiatry, OkayamaUniversity HospitalYosukeMatsumotoOkayama University Hospital Gender CenterManabuTakakiDepartment of Neuropsychiatry, OkayamaUniversity Faculty of Medicine, Dentistry andPharmaceutical SciencesBackground: Inattention due to attention-deficit/hyperactivity disorder (ADHD) can lead to forgetfulness. Transient epileptic amnesia (TEA) can cause forgetfulness, similar to ADHD. We report a patient with ADHD who developed TEA.<br>
Case Presentation: The patient was a 40-year-old woman with ADHD. She has been prone to forgetfulness since childhood. Two years before visiting our outpatient clinic, she had begun to occasionally forget events that had occurred several days earlier. However, she was largely unaware of the emergence of new amnestic symptoms. She had also begun to experience various other amnestic symptoms 2 months before she visited our clinic, which prompted her to visit our outpatient clinic. The combination of a detailed interview, electroencephalography (EEG) examination, and consideration of TEA enabled us to diagnose her with TEA and provide treatment accordingly. In our patient, daily forgetfulness due to ADHD delayed the recognition of new additional forgetfulness attributed to TEA.<br>
Conclusion: Psychiatrists need to consider TEA when patients with ADHD present with changes in or exacerbation of forgetfulness. We report a patient with ADHD who developed TEA. In our patient, daily forgetfulness due to ADHD delayed the recognition of new additional forgetfulness attributed to TEA. Psychiatrists need to consider TEA when patients with ADHD present with changes or exacerbation of forgetfulness. No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7842024Middle-Ear Salivary Gland Choristoma with Congenital, Single-Sided Hearing Loss349355ENYuichiroTominagaDepartment of Otolaryngology, Head and Neck Surgery, Hiroshima City, Hiroshima Citizens HospitalAkikoSugayaDepartment of Otolaryngology, Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShinKariyaDepartment of Otolaryngology, Head and Neck Surgery, Kawasaki Medical School HospitalAikoShimizuDepartment of Otolaryngology, Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYukoKataokaDepartment of Otolaryngology, Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMizuoAndoDepartment of Otolaryngology, Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/67554Middle-ear salivary gland choristoma (SGCh) is a rare, benign tumor that causes conductive hearing loss owing to middle-ear morphological abnormalities. Early diagnosis is challenging, and surgical resection is indispensable for a definitive diagnosis. We report the case of a 3-year-old boy diagnosed with middle-ear SGCh during the follow-up period for left-sided hearing loss discovered at newborn hearing screening (NHS). Long-term follow-up after the NHS result, subsequent computed tomography/magnetic resonance imaging, and surgical resection led to its relatively early diagnosis and treatment.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7842024Surgical Treatment of an Abdominal Aortic Aneurysm in a Patient with a Liver Abscess345347ENGentaroKatoDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalYutoNarumiyaDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalMichihiroOkuyamaDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalShujiShimizuDepartment of Cardiovascular Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences and Okayama University HospitalKenjiSangawaDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalShuYamamotoDepartment of Cardiovascular Surgery, Kagawa Prefectural Central HospitalCase Report10.18926/AMO/67553The presence of an intraperitoneal source of infection, e.g., a liver abscess, can be an obstacle to performing an abdominal aortic surgery with a midline laparotomy because graft infection is one of the most critical complications of aortic surgery. We report the successful Y-grafting of a pararenal abdominal aortic aneurysm through a retroperitoneal approach in a 67-year-old male undergoing liver abscess drainage. The retroperitoneal approach to the abdominal aorta may be useful for abdominal aortic surgery in patients with a localized intraperitoneal infection.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7842024Pneumocephalus with Inverted Papilloma in the Frontoethmoidal Sinus: Case Report and Literature Review337343ENSeiichiroMakiharaDepartment of Otolaryngology-Head & Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYoshihiroOtaniDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesKensukeUraguchiDepartment of Otolaryngology-Head & Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesSawakoOnoDepartment of Pathology and Oncology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesAikoShimizuDepartment of Otolaryngology-Head & Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesRyosukeIkemachiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesYosukeOkazakiDepartment of Neurological Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesTomoyukiOtaDepartment of Plastic and Reconstructive Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesHiroshiMatsumotoDepartment of Plastic and Reconstructive Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesShotaroMiyamotoDepartment of Otolaryngology-Head & Neck Surgery, Kagawa Rosai HospitalMunechikaTsumuraDepartment of Otolaryngology-Head & Neck Surgery, Kagawa Rosai HospitalSeiyaHayashiDepartment of Neurosurgery, Kagawa Rosai HospitalMichiariUmakoshiDepartment of Neurosurgery, Kagawa Rosai HospitalKojiHirashitaDepartment of Neurosurgery, Kagawa Rosai HospitalMizuoAndoDepartment of Otolaryngology-Head & Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/67550Here, we describe the unique case of a pneumocephalus originating from an inverted papilloma (IP) in the frontoethmoidal sinus. A 71-year-old man with diabetes presented with headaches and altered consciousness. Imaging revealed the pneumocephalus together with bone destruction in the left frontal sinus. He underwent simultaneous endoscopic endonasal and transcranial surgery using an ORBEYE exoscope. Pathological diagnosis of the tumor confirmed IP. Post-surgery, the pneumocephalus was significantly resolved and the squamous cell carcinoma antigen level, which had been elevated, decreased. This case underscores the importance of a multidisciplinary approach and innovative surgical methods in treating complex sinonasal pathologies.No potential conflict of interest relevant to this article was reported.Okayama University Medical SchoolActa Medica Okayama0386-300X7842024A Rare Subglottic Pleomorphic Adenoma: Magnetic Resonance Findings331335ENChiekoFurukawaDepartment of Otolaryngology Head and Neck Surgery, Japanese Red Cross Society Himeji HospitalTomoyasuTachibanaDepartment of Otolaryngology Head and Neck Surgery, Japanese Red Cross Society Himeji HospitalTetsujiNobuhisaDepartment of Surgery, Japanese Red Cross Society Himeji HospitalYuichiroKanieDepartment of Radiology, Japanese Red Cross Society Himeji HospitalYojiWaniDepartment of Pathology, Japanese Red Cross Society Himeji HospitalJun-YaMatsumotoDepartment of Otolaryngology Head and Neck Surgery, Japanese Red Cross Society Himeji HospitalAkifumiKariyaDepartment of Otolaryngology Head and Neck Surgery, Japanese Red Cross Society Himeji HospitalAsukaSatoDepartment of Otolaryngology Head and Neck Surgery, Japanese Red Cross Society Himeji HospitalIichiroIshikawaDepartment of Surgery, Japanese Red Cross Society Himeji HospitalYutoNaoiDepartment of Otolaryngology Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesMizuoAndoDepartment of Otolaryngology Head and Neck Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesCase Report10.18926/AMO/67549No previous study has published magnetic resonance imaging (MRI) findings for a subglottic pleomorphic adenoma. Here, we describe the case of a 62-year-old man with a subglottic pleomorphic adenoma. Endoscopic findings revealed a smooth-surfaced tumor arising from the subglottic posterior wall. MRI revealed the lesion as an isointense region on T1-weighted images, which was homogeneously enhanced. This lesion showed a heterogeneously hyperintense region on T2-weighted images. Diffusion-weighted imaging (DWI) showed slightly high intensity in the same area, with a normal or only slightly high apparent diffusion coefficient (ADC). Laryngomicrosurgery was performed for transoral excision of the subglottic tumor, resulting in a postsurgical diagnosis of pleomorphic adenoma.No potential conflict of interest relevant to this article was reported.WileyActa Medica Okayama2577-171X642023A case of mucosal-associated lymphoid tissue lymphoma of the urachus253256ENKazumaTsuboiDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalKensukeBekkuDepartment of Urology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical SciencesKoheiHaisaDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalYutaKajiharaDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalTakujiTsugawaDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalYosukeInoueDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalTomokoSakoDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalWataruMuraoDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalShinEbaraDepartment of Urology, Hiroshima City Hiroshima Citizens HospitalIntroduction: Urachus carcinoma is a rare malignancy with an aggressive potential and a poor prognosis, and evidence is limited for its diagnosis and treatment.<br>
Case presentation: A 75-year-old man underwent fluorodeoxyglucose positron emission tomography/computed tomography for staging prostate cancer, and a mass (standardized uptake value max 9.5) was observed on the outside of the urinary bladder dome. T2-weighted magnetic resonance imaging showed the urachus and a low-intensity tumor, which suggested a malignant tumor. We suspected urachal carcinoma and performed total resection of the urachus and partial cystectomy. Pathological examination revealed mucosa-associated lymphoid tissue lymphoma with cells positive for CD20 and negative for CD3, CD5, and cyclin D1. After the surgery, no recurrence has been observed for more than 2 years.<br>
Conclusion: We encountered an extremely rare case of mucosa-associated lymphoid tissue lymphoma of the urachus. Surgical resection of the tumor provided an accurate diagnosis and good disease control.No potential conflict of interest relevant to this article was reported.Nature PortfolioActa Medica Okayama2045-23221412024Training high-performance deep learning classifier for diagnosis in oral cytology using diverse annotations17591ENShintaroSukegawaDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityFutaTanakaDepartment of Electrical, Electronic and Computer Engineering, Faculty of Engineering, Gifu UniversityKeisukeNakanoDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityTakeshiHaraDepartment of Electrical, Electronic and Computer Engineering, Faculty of Engineering, Gifu UniversityTakanagaOchiaiDivision of Oral Pathogenesis and Disease Control, Department of Oral Pathology, Asahi University School of DentistryKatsumitsuShimadaDepartment of Oral Pathology, Graduate School of Oral Medicine, Matsumoto Dental UniversityYutaInoueDepartment of Electrical, Electronic and Computer Engineering, Faculty of Engineering, Gifu UniversityYoshihiroTakiDepartment of Electrical, Electronic and Computer Engineering, Faculty of Engineering, Gifu UniversityFumiNakaiDepartment of Oral and Maxillofacial Surgery, Kagawa University Faculty of MedicineYasuhiroNakaiDepartment of Oral and Maxillofacial Surgery, Kagawa University Faculty of MedicineTakanoriIshihamaDepartment of Oral and Maxillofacial Surgery, Kagawa University Faculty of MedicineRyoMiyazakiStony Brook Cancer Center, Stony Brook UniversitySatoshiMurakamiDepartment of Oral Pathology, Graduate School of Oral Medicine, Matsumoto Dental UniversityHitoshiNagatsukaDepartment of Oral Pathology and Medicine, Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama UniversityMinoruMiyakeDepartment of Oral and Maxillofacial Surgery, Kagawa University Faculty of MedicineThe uncertainty of true labels in medical images hinders diagnosis owing to the variability across professionals when applying deep learning models. We used deep learning to obtain an optimal convolutional neural network (CNN) by adequately annotating data for oral exfoliative cytology considering labels from multiple oral pathologists. Six whole-slide images were processed using QuPath for segmenting them into tiles. The images were labeled by three oral pathologists, resulting in 14,535 images with the corresponding pathologists' annotations. Data from three pathologists who provided the same diagnosis were labeled as ground truth (GT) and used for testing. We investigated six models trained using the annotations of (1) pathologist A, (2) pathologist B, (3) pathologist C, (4) GT, (5) majority voting, and (6) a probabilistic model. We divided the test by cross-validation per slide dataset and examined the classification performance of the CNN with a ResNet50 baseline. Statistical evaluation was performed repeatedly and independently using every slide 10 times as test data. For the area under the curve, three cases showed the highest values (0.861, 0.955, and 0.991) for the probabilistic model. Regarding accuracy, two cases showed the highest values (0.988 and 0.967). For the models using the pathologists and GT annotations, many slides showed very low accuracy and large variations across tests. Hence, the classifier trained with probabilistic labels provided the optimal CNN for oral exfoliative cytology considering diagnoses from multiple pathologists. These results may lead to trusted medical artificial intelligence solutions that reflect diverse diagnoses of various professionals.No potential conflict of interest relevant to this article was reported.Springer Science and Business Media LLCActa Medica Okayama1471-24152412024Pathological findings in enucleated eyes of patients with neurofibromatosis type 1: report of a case with 15-year follow-up and review of 14 patients in the literature341ENToshihikoMatsuoGraduate School of Interdisciplinary Science and Engineering in Health Systems, Okayama UniversityKenjiNishidaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTakehiroTanakaDepartment of Pathology, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityTakayaSenoDepartment of Plastic and Reconstructive Surgery, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityKiyoshiYamadaDepartment of Plastic and Reconstructive Surgery, Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama UniversityShigekiOnoDepartment of Neurological Surgery, General Medical Center, Kawasaki Medical SchoolBackgrounds Iris nodules are frequently noted as clinical manifestations of neurofibromatosis type 1 but the other intraocular manifestations are rare. The purpose of this study is to present a patient with a phthisic eye who underwent enucleation for a cosmetic reason after 15-year follow-up and also to review 14 patients with enucleation described in the literature.<br>
Case presentation A 17-year-old man with neurofibromatosis type 1 from infancy underwent the enucleation of phthisic left eye and also had the resection of eyelid subcutaneous mass lesions on the left side for a cosmetic reason. He had undergone four-time preceding surgeries for eyelid and orbital mass reduction on the left side in childhood and had developed total retinal detachment 10 years previously. Pathologically, the enucleated eye showed massive retinal gliosis positive for both S-100 and glial fibrillary acidic protein (GFAP) in the area with involvement of the detached retinal neuronal layer, together with a more fibrotic lesion along the choroid which were, in contrast, negative for both S-100 and GFAP. The choroid, ciliary body, and iris did not show apparent neurofibroma while episcleral neurofibroma was present.<br>
Literature review In review of enucleated eyes of 14 patients in the literature, buphthalmic eyes with early-onset glaucoma on the unilateral side was clinically diagnosed in 9 patients who frequently showed varying extent of hemifacial neurofibromatosis which involved the eyelid and orbit on the same side. Pathologically, neurofibromas in varying extent were found in the choroid of 12 patients. One patient showed choroidal malignant melanoma on the left side and fusiform enlargement of the optic nerve on the right side suspected of optic nerve glioma. The phthisic eye in another patient showed massive retinal gliosis similar to the present patient.<br>
Conclusions In summary of the 15 patients with neurofibromatosis type 1, including the present patient, buphthalmic or phthisic eyes with no vision were enucleated for cosmetic reasons and showed choroidal neurofibroma in most patients and massive retinal gliosis in two patients including the present patient.No potential conflict of interest relevant to this article was reported.SpringerActa Medica Okayama2198-40341012024Long-term follow-up of a patient with Parkinson's disease under nursing care after replacement of fixed implant-supported prostheses with an implant overdenture: a case report37ENKanaTokumotoDepartment of Oral and Maxillofacial Surgery, School of Medicine, Hyogo Medical UniversityTakuyaMinoOkayama University Dental SchoolIkueTosaOkayama University Dental SchoolKoOmoriDepartment of Oral Rehabilitation and Regenerative Medicine, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of MedicineMichiyoYamamotoDental Clinic, AINOSATO ClinicKazukiTakaokaDepartment of Oral and Maxillofacial Surgery, Shiga University of Medical ScienceKenjiMaekawaDepartment of Removable Prosthodontics and Occlusion, Osaka Dental UniversityTakuoKubokiOkayama University Dental SchoolHiromitsuKishimotoDepartment of Oral and Maxillofacial Surgery, School of Medicine, Hyogo Medical UniversityBackground In older patients with progressive neurodegeneration, replacing fixed implant-supported prostheses (FIP) with implant overdentures (IOD) has been proposed to prevent future mucosal injury and create an oral environment that is easier for caregivers to clean. However, there have been no reports on the progress after replacing FIP with IOD. In this report, we present the progress of an older patient with Parkinson’s disease in whom FIP was replaced with IOD.<br>
Case presentation An 81-year-old male patient with Parkinson’s disease presented to our outpatient clinic with bruxism and crossbites. FIPs, with five Brånemark system implants, were placed in the bilateral lower molars. The FIP was replaced with an IOD with two locator attachments to create an oral environment that was easier for caregivers to clean and allow easy recovery of masticatory function if residual teeth were fractured in the care environment. As his systemic condition deteriorated, treatment was changed from outpatient to in-home visits. During dental care visits, professional oral cleaning and denture repair were continued, and good nutritional status was maintained. However, the patient developed cholecystitis and was hospitalized. During hospitalization, gastrostomy was performed because he developed aspiration pneumonia. After discharge from the hospital, the patient remained in bed all day and could not wear an IOD, resulting in buccal mucosa ulceration due to abrasion of the locator abutment. We decided to replace the abutment with cover screws; however, not all the implants could sleep submucosally. Although regular oral cleaning was resumed, new ulcers developed even when cover screws were installed. Additionally, swelling and drainage were observed at the peri-implant mucosal site where peri-implantitis had once occurred during an outpatient visit. The patient was readmitted to the hospital for a urinary tract infection, and subsequent visits were abandoned.<br>
Conclusions By replacing FIP with IOD in an older patient with Parkinson’s disease, we addressed a barrier to caregiver-provided oral management. The removable prosthesis facilitated smooth oral care by caregivers and functional recovery in the event of trouble with residual teeth. However, it could not completely avoid the recurrence of buccal mucosal ulcers or peri-implantitis.No potential conflict of interest relevant to this article was reported.