This is a case of remarkable intracranial extramedullary hematopoiesis (EMH) in a 63-year-old male patient with a history of idiopathic myelofibrosis. Intracranial EMH was suspected following cranial computed tomography and magnetic resonance imaging and was found in the same sites as the bilateral subdural hematomas. The patient subsequently received whole brain irradiation (30 Gy). The lesion was controlled for one and a half years. After the patient died from bacterial infection, intracranial EMH was confirmed in autopsy and histological diagnoses.

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