Diffuse cutaneous leishmaniasis (DCL) is an unusual form of cutaneous leishmaniasis mainly caused by infection with Leishmania aethiopica in the Old World. In this paper, diffuse cutaneous leishmaniasis was reported for the first time, in an Egyptian patient from Sinai Peninsula resulting from infection with L. major zymodeme LOND-1 as proved by enzymatic characterization, using seven enzymes.