Recurrence of visceral and muco-cutaneous leishmaniasis in a patient under immunosuppressive therapy

Abstract

BackgroundLeishmaniasis is a protozoan disease caused by parasites of the genus Leishmania, transmitted to humans by sandflies. The diagnosis of leishmaniasis is often challenging as it mimics many other infectious or malignant diseases. The disease can present in three ways: cutaneous, mucocutaneous, or visceral leishmaniasis, which rarely occur together or consecutively.Case presentationThe patient was a 52 years old immunosuppressed Belgian woman with a long history of severe rheumatoid arthritis. She underwent bone marrow biopsy to explore thrombocytopenia. Diagnosis of visceral leishmaniasis was made by identification of Leishman Donovan (LD) bodies in macrophages. Treatment with liposomal amphotericin B was successful. She later developed cutaneous leishmaniasis treated with amphotericin B lipid complex. She next presented with relapsing cutaneous lesions followed by rapidly progressing lymphadenopathies. Biopsy confirmed the diagnosis of leishmaniasis. Treatments by miltefosine, amphotericin B, N-methyl-glucamine antimoniate were subsequently initiated. She later presented a recurrent bone marrow involvement treated with intramuscular paromomycin and miltefosine. She died two years later from leukemia. At the time of death, she presented with a mucosal destruction of the nose. A Leishmania-specific PCR (Polymerase Chain Reaction) identified L. infantum as etiological agent.ConclusionsClinicians should be aware of the potential concomitant or sequential involvement of multiple anatomic localizations of Leishmania in immunosuppressed patients.

A comprehensive systematic review of leishmaniasis in patients undergoing drug-induced immunosuppression for the treatment of dermatological, rheumatological and gastroenterological diseases

“…Considering the clinical picture, only 39 atypical leishmaniasis cases were reported. A Leishmania species known to cause visceral disease (Leishmania infantum) resulted in mucocutaneous leishmaniasis in only one case 10 .…”

Visceral Dissemination of Mucocutaneous Leishmaniasis in a Kidney Transplant Recipient

“…Leishmaniasis in the Mediterranean basin, is a zoonotic disease mostly caused by Leishmania infantum, usually associated with VL and less frequently with CL [6]. In 2009 there had been an outbreak of leishmaniasis in Madrid (Spain), with more than 700 cases declared, mainly presenting with the cutaneous form of the disease [7].…”

“…Clinical manifestations of the disease are mostly based on three different syndromes: Visceral leishmaniasis (VL), cutaneous (CL), or mucocutaneous (MCL) [ 1 , 2 , 3 , 5 ]. Leishmaniasis in the Mediterranean basin, is a zoonotic disease mostly caused by Leishmania infantum , usually associated with VL and less frequently with CL [ 6 ]. In 2009 there had been an outbreak of leishmaniasis in Madrid (Spain), with more than 700 cases declared, mainly presenting with the cutaneous form of the disease [ 7 ].…”

Secondary Prophylaxis with Liposomal Amphotericin B in a Patient with Mucosal Leishmaniasis Undergoing Immunobiological Therapy for Active Ankylosing Spondylitis

“…This situation becomes critical in the presence of comorbidities such as HIV infection, immunosuppressive therapy for cancer and autoimmune disorders, and immunosuppression following organ transplantation that alter the patient's immune response, thereby compromising therapeutic response and evolution of mucosal tissue. 10,11 Tumor necrosis factor inhibitors have revolutionized the treatment of ankylosing spondylitis (AS), and five of them have been shown to be useful in the control of AS, namely, infliximab, etanercept, adalimumab, certolizumab, and golimumab. All these TNF inhibitors can significantly improve function, spinal mobility, peripheral synovitis, enthesitis, and quality of life scores.…”