BACKGROUNDEpidemiological data related to leishmaniases orLeishmaniainfection in horses are scarce. However, studies carried out in different regions in the world showed equids parasitised byLeishmania braziliensis,L. infantumandL. martiniquensis.OBJECTIVESIdentify theLeishmaniaspecies causing cutaneous leishmaniasis in a mare, living in Rio de Janeiro State (Brazil), and search the presence ofLeishmaniaviruses in the isolated parasite.METHODSIsoenzymes and polymerase chain reaction (PCR) targeting ITSrDNA region followed by sequencing were conducted for typing the isolated parasite. A search forLeishmaniavirus infection was also performed.FINDINGSThe mare presented skin nodules and ulcers in the left pinna caused byLeishmaniaspp. that was detected by culture and PCR. The parasite was identified asLeishmania(Mundinia)martiniquensis, infected byLeishbunyavirus(LBV), representing the first description of this species in South America. The animal travelled to different Brazilian regions, but not to outside the country.MAIN CONCLUSIONSThe worldwide distribution ofL.martiniquensisand its infection by LBV were confirmed in this study, indicating the autochthonous transmission cycle in Brazil. The clinical profile of the disease in the mare, showing fast spontaneous healing of cutaneous lesions, may indicate that skin lesions related toL. martiniquensisinfection in horses might be underdiagnosed.