start-ver=1.4 cd-journal=joma no-vol=100 cd-vols= no-issue=13 article-no= start-page=e25265 end-page= dt-received= dt-revised= dt-accepted= dt-pub-year=2021 dt-pub=20210402 dt-online= en-article= kn-article= en-subject= kn-subject= en-title= kn-title=Disseminated gonococcal infection in a Japanese man with complement 7 deficiency with compound heterozygous variants A case report en-subtitle= kn-subtitle= en-abstract= kn-abstract=Rationale: Complement deficiency are known to be predisposed to disseminated gonococcal infection (DGI). We herein present a case of DGI involving a Japanese man who latently had a complement 7 deficiency with compound heterozygous variants.
Patient concerns: A previously healthy 51-year-old Japanese man complained of sudden-onset high fever. Physical examination revealed various skin lesions including red papules on his trunk and extremities, an impetigo-like pustule on left forearm, and tendinitis of his right forefinger.
Diagnosis: Blood culture testing detected gram-negative cocci, which was confirmed to be Neisseria gonorrhoeae based on mass spectrometry and a pathogen-specific PCR test.
Interventions: Screening tests for underlying immunocompromised factors uncovered that complement activities (CH50) was undetectable. With a suspicion of a congenital complement deficiency, genetic analysis revealed rare single nucleotide variants in complement 7 (C7), including c.281-1G>T and a novel variant c.1454C>T (p.A485V). CH50 was normally recovered by adding purified human C7 to the patient's serum, supporting that the patient has C7 deficiency with compound heterozygous variants.
Outcomes: Under a diagnosis of DGI, the patient underwent an antibiotic treatment with cefotaxime for a week and was discharged without any sequela.
Lessons: DGI is a rare sexually-transmitted infection that potentially induces systemic complications. Complement immunity usually defeats N. gonorrhoeae and prevents the organism from causing DGI. This case highlighted the importance of suspecting a complement deficiency when a person develops DGI. en-copyright= kn-copyright= en-aut-name=KageyamaMisaki en-aut-sei=Kageyama en-aut-mei=Misaki kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=1 ORCID= en-aut-name=HagiyaHideharu en-aut-sei=Hagiya en-aut-mei=Hideharu kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=2 ORCID= en-aut-name=UedaYasutaka en-aut-sei=Ueda en-aut-mei=Yasutaka kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=3 ORCID= en-aut-name=OhtaniKatsuki en-aut-sei=Ohtani en-aut-mei=Katsuki kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=4 ORCID= en-aut-name=FukumoriYasuo en-aut-sei=Fukumori en-aut-mei=Yasuo kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=5 ORCID= en-aut-name=InoueNorimitsu en-aut-sei=Inoue en-aut-mei=Norimitsu kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=6 ORCID= en-aut-name=WakamiyaNobutaka en-aut-sei=Wakamiya en-aut-mei=Nobutaka kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=7 ORCID= en-aut-name=YonedaNanoka en-aut-sei=Yoneda en-aut-mei=Nanoka kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=8 ORCID= en-aut-name=KimuraKeigo en-aut-sei=Kimura en-aut-mei=Keigo kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=9 ORCID= en-aut-name=NagasawaMotonori en-aut-sei=Nagasawa en-aut-mei=Motonori kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=10 ORCID= en-aut-name=NakagamiFutoshi en-aut-sei=Nakagami en-aut-mei=Futoshi kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=11 ORCID= en-aut-name=NishiIsao en-aut-sei=Nishi en-aut-mei=Isao kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=12 ORCID= en-aut-name=SugimotoKen en-aut-sei=Sugimoto en-aut-mei=Ken kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=13 ORCID= en-aut-name=RakugiHiromi en-aut-sei=Rakugi en-aut-mei=Hiromi kn-aut-name= kn-aut-sei= kn-aut-mei= aut-affil-num=14 ORCID= affil-num=1 en-affil=Department of General Medicine, Osaka University Hospital kn-affil= affil-num=2 en-affil=Department of General Medicine, Osaka University Hospital kn-affil= affil-num=3 en-affil=Department of Hematology and Oncology, Osaka University Hospital kn-affil= affil-num=4 en-affil=The Japanese Association for Complement Research kn-affil= affil-num=5 en-affil=Department of Molecular Genetics, Wakayama Medical University kn-affil= affil-num=6 en-affil=The Japanese Association for Complement Research kn-affil= affil-num=7 en-affil=The Japanese Association for Complement Research kn-affil= affil-num=8 en-affil=Laboratory for Clinical Investigation, Osaka University Hospital kn-affil= affil-num=9 en-affil=Laboratory for Clinical Investigation, Osaka University Hospital kn-affil= affil-num=10 en-affil=Department of General Medicine, Osaka University Hospital kn-affil= affil-num=11 en-affil=Department of General Medicine, Osaka University Hospital kn-affil= affil-num=12 en-affil=Laboratory for Clinical Investigation, Osaka University Hospital kn-affil= affil-num=13 en-affil=Department of General Medicine, Osaka University Hospital kn-affil= affil-num=14 en-affil=Department of General Medicine, Osaka University Hospital kn-affil= en-keyword=complement addition test kn-keyword=complement addition test en-keyword=complement deficiency kn-keyword=complement deficiency en-keyword=disseminated gonococcal infection kn-keyword=disseminated gonococcal infection en-keyword=genome analysis kn-keyword=genome analysis en-keyword=Neisseria gonorrhoeae kn-keyword=Neisseria gonorrhoeae en-keyword=sexually transmitted infection kn-keyword=sexually transmitted infection END