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Gata-Haydarpaşa Eğitim Hastanesi, Hematoloji-Onkoloji Servisi, Ankara We report a case of mast cell leukemia. The leukemia variant of systemic mast cell disease is rapidly fatal in contrast to the non leukemic cases, which follow an indolent clinical course. On the basis of our case and previously reported cases, mast cell leukemia is characterized by a substantial increase in atypical mast cell in the peripheral blood, diffuse infiltration with atypical mast cells in the bone marrow, a strong association with peptic ulcer disease, prominent constitutional symptoms and hepatosplenomegaly. These cases should be distinguished from malignant mastocytosis and also from, cases of acute non lymphocytic leukemia that arise in the background of systemic mast cell disease. There is no established treatment modality in mast cell leukemia. Keywords :