The first known case of Blastoschizomyces capitatus meningitis occurring in an allogeneic bone marrow recipient on steroid and cyclosporine therapy for chronic graft-versus-host disease is reported. An 11-month course of treatment with oral fluconazole resulted in resolution of the meningeal syndrome and eradication of Blastoschizomyces capitatus from the cerebrospinal fluid. Three months after discontinuation of fluconazole the patient died due to idiopathic interstitial pneumonia and bilateral pneumothorax, without clinical signs of meningitis. Post mortem examination showed meningeal fungus invasion consistent with Blastoschizomyces capitatus infection. Oral fluconazole treatment thus did not eradicate the fungal infection, but achieved significant control of the meningitis on an outpatient basis.