To describe the first known case of Trichosporon Asahii meningitis in a patient with MOG antibody–associated myelitis and review relevant literature.

Trichosporon asahii is an emerging opportunistic yeast that causes invasive infections with high mortality, particularly in immunocompromised hosts. Central nervous system (CNS) dissemination is exceedingly rare, and its occurrence in patients with myelin oligodendrocyte glycoprotein-associated disease (MOGAD) has not been reported previously. 

A 44-year-old immunocompetent male presented with sudden onset fever, paraparesis, and bladder incontinence. MRI revealed T2/FLAIR hyperintensities at D12–L1, and serum MOG antibodies were positive, confirming MOG-associated myelitis. He improved with corticosteroids and azathioprine but re-presented 44 days later with headache and vomiting. CSF showed elevated protein, and fungal culture yielded T. asahii. The patient received intravenous followed by oral fluconazole, achieving complete recovery.