Ochroconis gallopavum is a potentially fatal dematiaceous fungus causing opportunistic infections in immunocompromised hosts. We report the first case of disseminated O. gallopavum infection in a 13-year-old renal transplant recipient, which involved the brain, lung and spleen. He was treated with amphotericin B, itraconazole and voriconazole, a new antifungal agent first used to treat such an infection. Besides antifungal treatment, all immunosuppressive agents were stopped and automated peritoneal dialysis was resumed. The initial infection was under control with both clinical and radiological improvements after treatment. However, the patient later acquired Acremonium spp. peritonitis; he failed to respond to high-dose amphotericin B, and finally succumbed. A total of 13 reported O. gallopavum human infections, including the one described here, are reviewed. The most common site of involvement is the brain and the crude mortality rate is up to 46%. As the disease is potentially lethal in immunocompromised hosts, empirical antifungal coverage should be considered in post-renal transplant recipients with suspected brain abscess. Early biopsy of lesion for histopathological and microbiological diagnosis would be essential in managing such cases.