We describe here a case of disseminated cutaneous leishmaniasis due to Leishmania major in a severely immunocompromised patient from Burkino Faso, Africa, who is infected with human immunodeficiency virus-1. The skin lesions failed to respond to full treatment courses of amphotericin B, sodium stibogluconate, and liposomal amphotericin B but were successfully treated with miltefosine, an alkylphosphocholine analogue.