AbstractBackground Cutaneous leishmaniasis is widespread in the Yemen, but has not been fully documented.Methods We have studied 42 cases from the Hajjah and Amran Governorates of the Yemen Republic. The clinical profile of these cases was recorded in a special protocol. The diagnosis was based on positive slit skin smear and histopathologic studies when needed.Results The age of the patients ranged between 1 and 65 years (median age, 19 years) and the duration of the disease ranged between 2 months and 12 years (median duration, 6 months). Most of the patients had a single lesion. Slit skin smear was positive for parasites in 35 cases (highly positive in 15). From the analysis of features, the disease could be classified into dry type (31 patients), wet type (seven patients), and chronic cutaneous leishmaniasis (four patients). Most of the patients required prolonged (4–6 weeks) treatment with sodium stibogluconate; the smear was still positive in eight patients, seven of whom were further treated successfully with a combination of rifampicin and isoniazid for 2–3 months. One who was treated successfully with intralesional injections of sodium stibogluconate.Conclusions Cutaneous leishmaniasis in this region seems to be endemic, has a long chronic course, shows a highly positive smear even 1–6 years after onset, and gives an inadequate response to prolonged systemic therapy. Further epidemiologic studies and taxonomic differentiation of the species are required.