A 24-year-old man presented with cerebral aspergillus fungal granuloma involving the left frontal region secondary to pulmonary aspergillosis. He was otherwise healthy with no evidence of immune-suppression. Because of poor penetration of amphotericin B into the brain and cerebrospinal fluid (CSF), this patient was treated by a combination of systemic and local therapy in addition to surgical excision resulting in a cure with follow up for more than three years. This form of treatment produced no untoward long-term side effects or neurological sequel. On review of the literature on aspergillosis of the central nervous system (CNS), we found that six patients, including the present case, have been reported who survived longer than 1 year; their treatment included local administration of antifungal agent in the abscess cavity or into cerebral ventricles in order to control this devastating, treatment-resistant pathological fungal infection.