Extramedullary hematopoiesis is manifested by several clinical hematologic disorders including beta-thalassemia major and thalassemia intermedia. We report an unusual case of extramedullary hematopoiesis, which presented as a painful presacral mass in a patient with beta-thalassemia intermedia. The diagnosis was confirmed by a needle biopsy guided by computed tomography. The patient responded well to radiation therapy to the sacral region.