We herein report a case of bilateral frontal horn cysts. The infant was delivered with a low birth weight (1,710g) at 31 weeks, 0 days by emergency Cesarean section. She was severely asphyxiated and exhibited respiratory distress syndrome. Surfactant was administered, and mechanical ventilation was required until 21 days of age. Brain computed tomography (CT) at 45 days of age revealed bilateral cysts adjacent to the frontal horns of the lateral ventricles. Her growth and development were normal. At 1 and a half- years of age, she underwent brain CT again and the above-mentioned cystic abnormality had disappeared. No dilatation or irregularity of the lateral ventricles was found. Normal development and transient abnormal cystic findings in brain CT suggested a diagnosis of frontal horn cysts. Frontal horn cysts should be considered as the causes of cystic lesions of the brain.