OBJECTIVE AND IMPORTANCEOnly a few cerebral infections with the dark-walled moldRamichloridium obovoideum(Ramichloridium mackenziei) have been reported in the literature. Central nervous system infections caused by this fungus have poor prognoses; the optimal medical and surgical treatments have not yet been established. We report a case of cerebralR. obovoideuminfection for which a combination of medical and surgical treatments failed.CLINICAL PRESENTATIONA 58-year-old Kuwaiti woman, with a history of chronic renal failure requiring hemodialysis, presented with a 3-day history of left frontal headache, blurry vision, dizziness, and right-sided clumsiness. Computed tomography demonstrated multiple, ring-enhancing, cerebral lesions (the largest of which measured 2–3 cm) in the deep left parieto-occipital region.INTERVENTIONA computed tomography-guided needle biopsy of the parieto-occipital lesion yielded 10 ml of dark caseous fluid. Stains demonstrated long, branching, septate hyphae. Fungal cultures grewR. obovoideum. The patient was treated with a combination of amphotericin B and itraconazole. The condition of the patient continued to deteriorate, and stereotactic aspiration of the largest lesion was performed. Despite this approach, the lesion progressed and the patient died.CONCLUSIONR. obovoideumis being increasingly recognized as a cause of cerebral abscesses in patients residing in the Middle East. Prognoses are poor, and responses to antifungal therapy are generally short-lived. Until more effective therapies are found, the greatest chance for adequate treatment involves early recognition, prompt treatment with antifungal agents, and attempts at complete resection.