The clinical course of a patient with chronic granulocytic leukemia cutis associated with myeloid metaplasia is described. Cutaneous manifestations of leukemia appeared only at the sites of bone marrow aspiration and vena puncture. Radiotherapy to both spleen and skin and chemotherapy with busulfan failed to control spreading cutaneous lesions. Treatment with hydroxyurea was followed by a marked reduction in spleen size, a decrease in white cell count and healing of the skin lesions.