Cutaneous leishmaniasis is a parasitic infection prevalent in many areas. The parasitic agent is usually transmitted by Phlebotomous sp. Lip leishmaniasis is a type of cutaneous leishmaniasis, which has rarely been reported. In a retrospective study performed in Ahwaz, center of Khuzestan province, southwest of Iran, 2861 patients were detected during 1986-1996. Seventeen of these patients (0.59%) had lower or upper lip leishmaniasis. The patients, age ranged from 3 months to 40 years. Duration of infection was 2 to 4 months. Eleven patients had only lip lesions whereas 6 patients had one or more skin lesions elsewhere. The appearance of lesions differed from nodules to crusted ulcers, usually observed on the vermilion border. Most of the cases failed to respond to topical or systemic antibiotic therapy. The diagnosis was based on the demonstration of Leishman bodies in Giemsa stained smears.