The unusual appearance of extensive skin ulcerations has been reported in patients with chronic myelogenous leukemia (CML) undergoing continuous chemotherapy with hydroxyurea. It is thought that hydroxyurea, an antineoplastic agent with selective cytotoxicity for cells that divide most actively (such as those of the skin), causes these ulcerations through impairment of normal wound healing in areas of common trauma. The most common site of ulcers is the leg, where the ulcers are often extremely painful, with violaceous macules surrounding them, and are associated with extensive edema. On biopsy, histological vascular changes include leukocytoclastic vasculitis, perivascular lymphocytic infiltration, formation of thrombus, swelling of the endothelial cells, and thickening of the vascular walls. We report successful split-thickness skin grafting on hydroxyurea-related leg ulcers after preoperative discontinuation of hydroxyurea treatment in a patient with CML. The possible pathogenesis of hydroxyurea-related leg ulcers is discussed.