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

Abstract

Background: Leishmaniasis is a chronic protozoan disease in which organisms are found within phagolysosomes of the mononuclear phagocyte system. There are three major forms: cutaneous, mucocutaneous and visceral. We report the first case of visceral leishmaniasis with cutaneous involvement in a patient with rheumatoid arthritis treated with the anti-tumour necrosis factor (anti-TNF) adalimumab. Objective: To highlight cutaneous leishmaniasis as the first indicator of a kala-azar disease in a patient treated with anti-TNF and to review the literature on leishmaniasis in the context of anti-TNF therapy. Case Report: A 59-year-old woman presented with a crusted plaque on the right elbow 34 months after the initiation of adalimumab. A cutaneous biopsy showed intracellular amastigotes. No Leishmania parasites were observed in a bone marrow aspirate, but laboratory tests showed anaemia and impaired liver function, abdominal ultrasonography showed hepatomegaly, and ELISA serology was strongly positive for Leishmania antibodies in serum and urine. Adalimumab was withdrawn and treatment combining intralesional pentavalent antimonials and liposomal amphotericin was started. Eight weeks later, the leishmaniasis had resolved. Conclusion: A skin biopsy disclosing leishmaniasis should prompt tests to rule out visceral leishmaniasis, especially in an area such as the Mediterranean where the prevalence of latent Leishmania infection is high.

Tumor necrosis factor alpha neutralization has no direct effect on parasite burden, but causes impaired IFN-γ production by spleen cells from human visceral leishmaniasis patients

“…VL development and/or reactivation of leishmaniasis has been observed following TNF-α neutralizing therapy [20; 21; 22; 23; 24; 25; 26; 27]. Higher levels of TNF-α may exacerbate disease, and polymorphism in an allele associated with higher production of serum TNF-α has been linked to VL [28].…”

Cutaneous leishmaniasis mimicking dactylitis in a patient with rheumatoid arthritis treated with certolizumab

“…Leishmaniasis is a rare chronic protozoal disease that involves the mononuclear phagocytic system. Since 2004, 37 cases of patients diagnosed with leishmaniasis while on treatment with different types of biological anti‐tumour necrosis factor therapies have been reported . We present a case of an extraordinary form of cutaneous leishmaniasis in a patient treated with certolizumab, an association not previously reported, to our knowledge.…”

“…In addition, there was no visceral involvement despite basal immunosuppressive treatment and multiple cutaneous lesions. This is not a common finding, as anti‐TNF drugs are associated with a high risk of developing visceral leishmaniasis …”

Cutaneous leishmaniasis associated with TNF-α blockers: a case report

“…However, there are few reports of the reactivation of leishmaniasis after the use of this type of treatment 6. Traditionally, leishmaniasis affects the poorest populations as it is associated with malnutrition, population displacements, poor housing conditions, weakness of the immune system and lack of resources.…”