Cutaneous leishmaniasis (CL) in the Old World is caused mainly by three species of Leishmania: L. major, L. tropica and L. aethiopica, and sporadically by L. infantum and L. donovani. In Ethiopia, zoonotic cutaneous leishmaniasis, caused by L. aethiopica, is a major public health problem affecting thousands of people in the highlands. By contrast, little is known about the existence and epidemiology of CL due to L. tropica. In this report, we provide the first well-documented case of CL in Ethiopia caused by L. tropica. The patient acquired the infection in Awash valley of the Ethiopian Rift Valley (northeastern Ethiopia), where Phlebotomus sergenti and P. saevus have previously been found infected by L. tropica. Using the isoenzyme electrophoresis technique, the isolate was found to belong to a variant of L. tropica zymodeme MON-71, one of the new zymodemes found in Ethiopia from P. sergenti in the same region so far. The epidemiological implications of the finding are discussed.