A 30-year-old female presented with a rare case of isolated recurrence of granulocytic sarcoma manifesting as extra- and intracranial masses 16 months after successful treatment of acute myeloblastic leukemia (M-2). She presented with a swelling located on her forehead that had appeared just after hitting her forehead, and never diminished in size. The mass was elastic hard and not freely mobile. Computed tomography and magnetic resonance imaging demonstrated enhanced masses in the right frontal extra- and intracranial region with no bone destruction. There was no evidence of relapse in the bone marrow. Needle aspiration biopsy of the subscalpal mass was performed. Fluorescence in situ hybridization revealed AML1/MTG8 fusion gene associated with t(8; 21). Two courses of systemic chemotherapy with high-dose cytarabine and total neural axis irradiation resulted in complete remission.