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Abstract Cerebral phaeohyphomycosis is a fungal brain infection with a high fatality rate. It is caused by dematiaceous fungi and is increasingly recognized as a cause of serious illness in both immunocompetent and immunocompromised patients. We report cerebral phaeohyphomycosis in a liver transplant recipient. He was treated with multiple surgeries and antifungals and made a complete recovery. This report highlights that early and aggressive surgical intervention and extended antifungal coverage can have a positive outcome even in immunocompromised patients. The fungal infection in immunocompromised patients should be considered and treated aggressively.