An Atypical Presentation of Visceral Leishmaniasis Infection in a Patient With Rheumatoid Arthritis Treated With Infliximab

Abstract

Tumor necrosis factor alpha (TNF-alpha) is a cytokine, implicated in the pathogenesis of many inflammatory diseases, as well as in the immune-mediated response to infection, especially against intracellular pathogens. TNF-alpha antagonists have represented a revolution in the management of connective tissue diseases, such as rheumatoid arthritis. However, the use of these agents has been implicated with the emergence of a growing number of opportunistic infections. Here we report the case of a visceral Leishmaniasis in a 77-year-old woman who had been previously treated for rheumatoid arthritis with infliximab. The atypical presentation of this patient, previously treated with an anti-TNF-alpha biologic agent, where no splenomegaly or hepatomegaly was identified, is emphasized.

Anti-Tumour Necrosis Factor-Induced Visceral and Cutaneous Leishmaniasis: Case Report and Review of the Literature

“…Since then, 37 cases (including ours) of anti-TNF-induced leishmaniasis have been reported (online supplementary table 1, see www.karger.com/doi/10.1159/000370238) [15,16,17,18,19,20,21,22,23,24,25,26,27,28,29,30,31,32,33,34,35,36,37,38,39,40,41,42,43,44,45]. All patients were living in or had travelled to leishmaniasis-endemic areas.…”

Visceral Leishmaniasis Mimicking Systemic Lupus Erythematosus

“…VL may also present as a life-threatening opportunistic infection in patients with rheumatoid arthritis treated with TNF-antagonists or methotrexate. 1,10 Our patient fulfilled the revised classification criteria for SLE. 11 Hypergammaglobulinemia, ANA, anemia, leukocytopenia, and thrombocytopenia are all immunologic and hematological manifestations of SLE, but also of VL, and may explain misdiagnosis and diagnostic delay as in our patient.…”

“…1 We would like to emphasize another atypical presentation of this parasitic infection that may mimic systemic lupus erythematosus (SLE), 2 and report on an erroneous diagnosis of lupusrelated hemophagocytic syndrome (HS) in a patient with VL.…”

Clinical use of anti-TNF therapy and increased risk of infections

“…Clinically, two basic forms are recognized: cutaneous leishmaniasis, a disfiguring and stigmatizing disease, and visceral leishmaniasis or kala-azar, a life-threatening but treatable condition if diagnosed early. TNF-α has a major role in mediating host protection against visceral leishmaniasis,169 so the use of anti-TNF agents may potentially cause worsening or reactivation of latent infection 169. Four case reports of visceral leishmaniasis occurring in rheumatoid arthritis patients treated with infliximab have been published 169–172.…”

“…TNF-α has a major role in mediating host protection against visceral leishmaniasis,169 so the use of anti-TNF agents may potentially cause worsening or reactivation of latent infection 169. Four case reports of visceral leishmaniasis occurring in rheumatoid arthritis patients treated with infliximab have been published 169–172. Diagnosis is made by the detection of the parasite in bone marrow or blood, or by PCR assays performed on blood and bone marrow 171…”