Oxymetholone, an androgen analogue, is widely used in the treatment of Fanconi aplastic anemia (FAA). Androgen is known to cause cerebral venous thrombosis (CVT). A 9-year-old girl under monitoring with a diagnosis of FAA presented to our hospital with severe headache. She had been receiving oxymetholone therapy for 1 year. Neurological examination was normal. Superior sagittal sinus thrombosis was detected at magnetic resonance imagining (MRI) of the brain. The thrombosis was thought to be oxymetholone-related, and oxymetholone therapy was discontinued. The patient was started on enoxoparin therapy and the symptoms resolved. Control images 4 months later were normal. We think that the risk of venous thrombosis must be considered with administering oxymetholone therapy in patients with FAA. [Med-Science 2017; 6(3.000): 576-8]