Two patients presented with rare isolated recurrence of granulocytic sarcoma. A 29-year-old male presented with an extra- and intracranial mass 10 years after bone marrow transplantation for chronic myeloid leukemia. A 34-year-old female presented with an intracranial mass 3 years after complete remission of acute myeloid leukemia-M2a. Both patients underwent surgical resection. The first patient received adequate postoperative radiotherapy and chemotherapy, and achieved complete remission without evidence of diseases during the 6-month follow up. The second patient only received whole brain radiotherapy, failed to respond, and died of systemic leukemia later. These two cases demonstrate that neurosurgeons should pay attention to the occurrence of isolated recurrent granulocytic sarcoma, especially in patients with a history of hematologic neoplasm. Immediate pathological and cytogenetic diagnoses are essential.