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North Shore University Hospital, Division of Infectious Diseases, Manhasset, USA DOI : 10.5137/1019-5149.JTN .2907-10.1 Fungal infections of the central nervous system (CNS) are uncommon and occur mainly in immunocompromised patients. We describe a case of central nervous system aspergilloma without any evidence of systemic or paranasal foci in a diabetic but otherwise immunocompetent 71-year-old female treated successfully with surgical resection and medical therapy with voriconazole. Magnetic resonance imaging (MRI) after 6 months of voriconazole showed improvement and no evidence of residual or recurrent disease. Given its good CNS penetration, voriconazole along with surgical resection appears to be promising in treatment of these infections. Our case also demonstrates the importance of surgical intervention in the diagnosis and management of these atypical cases. Keywords : Aspergillus, Central nervous system, Voriconazole