A case of a 38-year-old male farmer with a brain abscess caused by Cladophialophora bantiana is described. He had a 2 year history of non-insulin-dependent diabetes and myelodysplastic syndrome. A cranial computed tomography scan demonstrated a hypodense ring lesion with peripheral oedema and a midline shift in the left frontal lobe. A darkly pigmented mould was isolated from the brain abscess. The isolate was identified as C. bantiana based on its morphological features and DNA sequence analysis. The patient was unresponsive to burr hole aspiration and irrigation, as well as liposomal amphotericin B infusion, and died after discharge from the hospital. This is believed to be the first case of a cerebral abscess due to C. bantiana in China.