BACKGROUND Cladophialophora carrionii was detected postoperatively in a cerebral space-occupying lesion of a patient who had undergone ABO-incompatible renal transplantation. The infection was successfully treated with oral terbinafine and itraconazole. CASE REPORT An otherwise healthy 46-year-old man underwent ABO-incompatible renal transplantation. Postoperatively, he was hemodynamically stable and the graft was functioning well. Within 2 weeks, the patient developed clinical depression, followed by seizures and left-side hemiparesis. There were no skin findings. Radiological investigation showed 2 space-occupying lesions in the brain parenchyma. The patient's condition improved after partial frontal lobectomy and microsurgical abscess evacuation, with a short course of liposomal amphotericin B and a combination of oral terbinafine and itraconazole. Microbiological examination of the pus showed growth of C. carrionii, which predominantly causes subcutaneous mycoses. CONCLUSIONS It is very rare for melanized fungal infections to cause an exclusively cerebral disease without any skin involvement. Furthermore, among established cases, C. carrionii is a very rarely detected pathogen.