Abstract
PURPOSE: Atovaquone is an alternative drug that is used for the prevention and treatment of Pneumocystis pneumonia when the first-line drug, sulfamethoxazole-trimethoprim (ST combination), cannot be used due to side effects. However, atovaquone is known to cause ocular side effects including oculomucocutaneous syndrome and vortex keratopathy. In this report, we describe a patient who developed bilateral white granular diffuse corneal opacity that extended from the corneal sub-epithelium to the stroma after continuous oral atovaquone administration for 14 months. OBSERVATIONS: The patient was a 15-year-old male with no prior ophthalmological or family medical history, but with a medical history of aplastic anemia treatment at our hospital's pediatric department. Examination showed bilateral diffuse white granular corneal opacity that extended from the sub-epithelium to the stroma, with no other abnormalities in the anterior and posterior segments of both eyes. CONCLUSIONS AND IMPORTANCE: We encountered a rare case of bilateral corneal opacity resulting from oral atovaquone administration. Regular long-term ophthalmological examinations are necessary for patients taking atovaquone.