A 41-year-old man complain of debility, had presented leukocytes of 1.58×109/L, hemoglobin of 77g/L and platelets of 47×109/L in peripheral blood. Most blasts showed morphologic changes same as acute megakaryocytic leukemia on bone marrow smear (panels a-b), and transmission electron microscopy also illustrated aberrant membrane demarcation or canalicular systems as megakaryocytes (panels c-d). Examinations of chromosome and molecular were normal. Flow cytometry demonstrated 67.6% of blasts positive for CD117 and GPA but negative for CD34, and 21.5% of blasts positive for CD34, CD117 and MPO. The patient was diagnosed as erythroleukemia and bone marrow transplantation was performed. This illustrates an interesting case of erythrocyte leukemia with morphologic changes mimicking megakaryocytic leukemia.