Background: Unlike visceral leishmaniasis, cutaneous leishmaniasis is uncommonly described in patients with human immunodeficiency virus infection. Diffuse cutaneous forms due to Leishmania infantum are always accompanied by visceral parasite infection.
Case report: We report a case of cutaneous leishmaniasis without visceral extension which was the inaugural sign of immunodeficiency virus infection. Polymerase chain reaction amplification of the genome was used to identify Leishmania infantum. Pentamidine (2 injections at the dose of 4 mg/kg, separated by one week) followed by maintenance therapy at the same dose every two weeks was given. Clinical cure was obtained after the initial injections and the intracellular parasite infestation had disappeared at the histology control.
Discussion: This case was unusual in that it was the inaugural sign of HIV infection. In addition, it is the first reported case of diffuse cutaneous leishmaniasis without visceral extension of Leishmania infantum in an HIV-infected patient. As no therapeutic consensus has been established, pentamidine would appear to be interesting for this population which also requires pneumocystosis prophylaxy.