A fungal infection of the brain of a 55-year-old male patient is reported. The lesion and involved fungus were located exclusively in the right medial temporo-parietal region. The patient was successfully treated with surgical resection of the lesion and antifungal chemotherapy. Few pathogenic dematiaceous fungi exhibit neurotropism and can cause primary infection in the central nervous system (CNS). The etiological agent is described as a Nodulisporium species. To date Nodulisporium has never been reported as an agent of CNS infection in humans