We report the case of an elderly woman who had been on hydroxyurea for long-standing widespread psoriasis. After approximately 5 years's treatment with hydroxyurea, she developed a symmetrical dermatomyositis-like eruption on her hands, together with bilateral leg ulceration. Although similar skin eruptions have been reported after long-term hydroxyurea treatment, all of the previous patients were being treated for myeloproliferative disorders. A dermatomyositis-like eruption has not previously been reported to occur as a consequence of hydroxyurea treatment for psoriasis. Its recognition is important to prevent unnecessary investigation or treatment withdrawal.