Cryptococcal infections of the central nervous system (CNS) are infrequent in immunocompetent hosts and usually present as meningitis. However, fungal masses called cryptococcoma can sometimes be formed. We report a case in which intraventricular cryptococcoma in an immunocompetent patient was completely cured using liposomal amphotericin B (L-AMB) and voriconazole (VRCZ). A 56-year-old previously healthy man was admitted to our hospital with fever, headache and bilateral hand tremor lasting over three weeks. Cerebrospinal fluid (CSF) studies on admission showed meningitis with a white blood cell count of 228 cells/μL: mononuclear leukocytes, 96%; polymorphonuclear leukocytes, 4%; proteins, 157 mg/dL; and glucose, 50 mg/dL. Magnetic resonance imaging (MRI) showed a small, homogeneously enhanced lesion in the right lateral ventricle, and 18F-fluoro-2-deoxy-D-glucose and 11C-methionine positron emission tomography revealed abnormal uptake corresponding to the lesion. To reach a definitive diagnosis, surgical excision of the lesion was performed. Histopathological examination of the specimen showed moderate lymphocyte infiltration and numerous fungal spores, and periodic acid-Schiff and Grocott staining revealed the presence of Cryptococcus neoformans (C.neoformans) within the granuloma, leading to a diagnosis of CNS cryptococcoma. The patient underwent treatment with intravenous L-AMB for 2 weeks and oral VRCZ for 10 months. CSF cultures were negative for C.neoformans and no recurrences were identified on MRI. CNS cryptococcoma is a rare infection that may occur in patients with no known history of immunosuppression. This pathology can be difficult to distinguish from brain tumor, so early pathological diagnosis from an excised specimen is very important. Furthermore, administration of L-AMB and VRCZ may be effective in treating cases of CNS cryptococcoma.

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