Introduction:
Visceral leishmaniasis is a well-recognized opportunistic infection in people living with HIV (PLHIV). Unlike adults, in children this infection is frequently associated with hemophagocytic lymphohistiocytosis (HLH). We report a case of HLH in HIV-positive person with visceral leishmaniasis.

Case description:
A 25-year-old man known living with HIV since 2013 was admitted to infectious diseases department in March 2017. His clinical examination was clear. His initial viral load was 630,000 copies/mm3 and CD4+ cells count was 12/mm3. No opportunistic infections were noted. The patient was started on antiretroviral therapy. During hospitalization, he developed fever, asthenia, rhinorrhea, and odynophagia. The diagnosis of HLH was retained because of pancytopenia, cytolysis, hyponatremia, high level of ferrinemia, and hemophagocytosis. Etiological investigations revealed positive Leishmania PCR. Also, Leishmania was detected in sternal puncture. Patient received meglumine antimoniate (glucantime) 20 mg/kg/day for 21 days with favorable outcomes. To prevent relapse, he received meglumine antimoniate 20 mg/kg/month as long as his CD4+ count was less than 100 cells/mm3. After 1-year follow up, no relapse was detected.

Conclusions:
Clinical and laboratory presentation of visceral leishmaniasis in PLHIV may differ from classic kala-azar. In our case, HLH was the reason for VL discovery.