Isolated extramedullary relapse of chronic myelogenous leukemia (CML) after allogeneic stem cell transplant is rare. A case is reported of a patient who developed a granulocytic sarcoma of the cavernous sinus 7 months after allogeneic transplant for CML. He presented with neurologic deficits, and a mass lesion was found on imaging. No evidence of hematologic relapse was identified by bone marrow histology or cytogenetics. A premortem diagnosis was not possible, and the patient died 2 months later of an intracerebral hemorrhage after receiving various therapies directed against a presumed infectious etiology. Granulocytic sarcoma should be considered in the differential diagnosis of mass lesions presenting after allogeneic transplant for CML, even if there is no evidence of medullary disease.