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Background Cutaneous leishmaniasis is the most common form of leishmaniasis caused by flagellate protozoa of the genus Leishmania transmitted by sand fly bites. Old World leishmaniasis is endemic in the Mediterranean Sea and the neighbouring countries. We believe, that this case is interesting by the fact that we had a very rear disease case that can be observed in nonendemic area. We present a case of a 22-year-old man with a cutaneous leishmaniasis in a localised form of ulcers on the right cheek and the right part of the neck. Histopathological examination showed diffuse dermal infiltrate predominantly of macrophages with admixture of few lymphocytes, eosinophils and plasma cells. In a very small number of macrophages amastigotes were seen. On their surface and occasionally extracellularly rod-shaped kinetoplasts were noticeable. It should be stressed that both clinical and laboratory data were not peculiar for this disease. Adults in endemic areas have stable immunity for protozoal infections. This made diagnostication and timely management of the disease very difficult. But clinical effect of drug therapy which is specific for cutaneous leishmaniasis treatment proved, in spite of the absence of ulcer soft tissues, blood and cerebrospinal puncture Leishmania, that our diagnosis was correct. The case, described by us, may be interesting for dermatologists, parasitologists, surgeons and other medical specialists. Because of higher rate of travel and work abroad increased number of sporadic cases of cutaneous leishmaniasis in non-endemic areas should be taken into account. Cutaneous leishmaniasis is a rare disease in Kazakhstan, especially in the north region. Because of higher rate of travel and work abroad increased number of sporadic cases of cutaneous leishmaniasis in non-endemic areas should be taken into account.