A cutaneous ulcer is a lesser known complication of hydroxyurea treatment. Out of 39 patients [18 polycythaemia vera (PV), 13 essential thrombocythaemia (ET), 4 chronic myeloid leukemia (CML), 4 undefined myeloproliferative diseases (MPD)] treated with hydroxyurea, 6 (4ET, 1PV, 1CML) developed a cutaneous ulcer during a period of less that 2 years' treatment. In all but one of the patients the ulcers were situated in the ankle region. At the time of onset of ulceration, none of them had extreme values in their peripheral blood counts. All had one or more of the predisposing factors such as minor trauma or mild varicosity. None of the patients had any alteration in arterial or venous circulation when examined by non-invasive means. No hyperviscosity was found as measured by capillary viscosimeter. The ulcers were cured in three patients without discontinuation of the drug. One patient later developed an ulcer on the other leg. The ulcers healed in two patients only after having stopped the hydroxyurea medication. One patient still had the ulcer when she succumbed to the underlying CML in transformation. In conclusion, cutaneous, ulceration of the leg is relatively common during hydroxyurea therapy. Predisposing factors are also involved in its development. Its healing does not necessarily require the discontinuation of the drug.