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Extramedullary hematopoiesis (EMH) is a compensatory process that supplements defective hematopoiesis and is frequently observed in myeloproliferative neoplasms (MPNs). Liver, spleen, and lymph nodes are the most common sites of EMH, whereas its occurrence in the intracranial space is extremely rare. We experienced a case of EMH which developed in cranial dura mater after irradiation against massive splenomegaly. As general condition of the patient did not allow us pathological examination, the diagnosis was confirmed by postmortem autopsy.