A 45-year-old Sudanese man from western Sudan presented with oronasal leishmaniasis of three years duration. He had no history of previous kala-azar or cutaneous leishmaniasis. The parasite isolated from the oral mucosa was characterized by isoenzymes using 12 enzymes and by polymerase chain reaction amplification of kinetoplast DNA using species-specific primers. The specific primers gave products indistinguishable from those of the Leishmania donovani complex. However, the isoenzyme profile showed a zymodeme pattern that was significantly different from the zymodemes previously reported in the Sudan and the Ethiopian region.