The effect of the antiprotozoal drug pentamidine on the ultrastructure of the amastigote stage of Leishmania tropica was studied. The most remarkable change introduced by the treatment occurred in the kinetoplast-mitochondrion complex. All mitochondria were extremely enlarged and most kinetoplasts disintegrated into a network of fine filaments. The flagellar pocket was frequently dilated and filled with various quantities of double-membrane bounded bodies ejected from the pocket wall. The multivesicular bodies and lipid droplets were enlarged. The number of ribosomal granules decreased. Many protozoa had an irregular shape and damaged internal structure. The drug-induced changes of L. tropica are discussed in relation to similar effects previously observed in trypanosomes.