We present a 43-year-old immunocompetent man who developed meningitis caused by Pseudallescheria boydii. The patient had no history of near drowning, trauma, steroid administration, operations or any other underlying systemic disease. He presented with intermittent fever associated with headache, bilateral eye pain, and vomiting. Progressive hydrocephalus was noted during the course of the disease. Cerebrospinal fluid (CSF) from the ventricular system allowed culture of the organism. Although the disease was diagnosed antemortemly, the patient died after antifungal treatment. This case is reported because of the unusual pathogen, unresponsiveness to amphotericin B combined with 5-fluocytocin, and immunocompetence of the patient without any predisposing factors.