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Neoscytalidium is a phytopathogen that is often found in plants and soil. It mostly leads to skin and nail infections, and invasive diseases of the sinuses, lung, and brain have been described mostly in immunocompromised patients. We report a case of a post-renal transplant patient who received anti-thymocyte globulin for induction immunosuppression. A month after her transplant, she presented with fever and new-onset seizures, and computed tomography revealed a brain abscess with mass effects and herniation. The patient underwent abscess drainage and craniectomy. The pathological findings showed filamentous septate hyphae. The surgical culture rapidly grew wool-like colonies with a black reverse on Sabouraud agar. Lactophenol cotton blue staining showing septate branched hyphae with one to two arthroconidia cells with flattened ends. The patient was given a combination of amphotericin B and voriconazole but unfortunately died ten days after the diagnosis. This case highlights Neoscytalidium as a cause of invasive fungal disease in immunocompromised patients that is difficult to treat and is often fatal, even when combined surgical and medical therapies are used as treatment modalities. • Neoscytalidium spp. is a rare human pathogenic fungal disease that most likely leads to superficial infection. • Immunocompromised host is at particularly higher risk of invasive disease. • This is the fifth case in the literature describing cerebral invasive disease caused by Neoscytalidium spp. • The present case further confirms the aggressiveness of the disease and refractoriness to antifungal therapy. • The diagnoses need to be entertained, especially in an immunocompromised patient and who have history of exposure to agricultural areas. [ABSTRACT FROM AUTHOR]