Visceral leishmaniasis and macrophagic activation syndrome in a patient with rheumatoid arthritis under treatment with adalimumab

Abstract

Background Visceral leishmaniasis is a protozoan infection usually asymptomatic, but can progress to fatal disease in immunocompromised hosts, especially in HIV patients. Visceral leishmaniasis is rare among patients under immunosuppressive therapies, and even more among patients under anti-TNF-α treatment, where only four cases have been described. Objective 1) To describe a patient with rheumatoid arthritis receiving adalimumab who developed fever, pancytopenia, splenomegaly, and extreme hyperferritinemia. 2) To perform a review of the published cases of visceral leishmaniasis and anti-TNF-α therapy, and cases of coexisting leishmaniasis and macrophagic activation syndrome by search in PubMed (period 1991–2008). Results Visceral leishmaniasis was established by bone marrow aspiration, and although there was no histological confirmation, according to HLH-2004 criteria, a secondary macrophagic activation syndrome was established. The patient had a favourable outcome. Conclusion We report herein the fifth case of visceral leishmaniasis in a patient under TNF-α therapy, and the first one, to our knowledge, presenting a consequent secondary macrophagic activation syndrome.