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Background: Extramedullary hematopoiesis refers to formation of hematopoietic tissue and it is a compensatory phenomen in hematologic disease like congenital hemolytic anemia, myelofibrosis, pernicious anemia & Bone marrow infiltration secondary to malignancies. It has been reported in many sites but the most common sites are liver, spleen & lymph node. Case Report: A 34-year-old man with intermediate B-thalassemia had chronic headache and progressive loss of visual function. In CT scan a lobulated, non homogenous hyperdense mass was seen in left occipital horn with massive surrounding edema. In histologic examination the presence of hematopoietic tissue in brain parenchyma was confirmed.