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

Abstract

Background: Leishmaniasis 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 presentation: The 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.

Conclusions: Clinicians should be aware of the potential concomitant or sequential involvement of multiple anatomic localizations of Leishmania in immunosuppressed patients.

Keywords: Cutaneous leishmaniasis; Immunosuppression; Microbiology; Mucosal leishmaniasis; Parasitology; Visceral leishmaniasis.

Fig. 1

Visceral and cutaneous leishmaniasis. a. Bone marrow aspirate: May Grunwald Giemsa (original magnification: ×1000) showing two macrophages with abundant cytoplasm containing innumerable Leishmania amastigotes. b. Spleen histology: High power view of the splenic red pulp comprising a prominent infiltrate of histiocytes and plasma cells; the histiocytes show numerous cytoplasmic punctate bodies surrounded by a clear halo, suggestive of Leishman bodies (hematoxylin eosin staining; original magnification: ×400). c. Cutaneous histology: Diffuse infiltrate in the upper dermis consisting of predominantly large histiocytes, plasma cells and a few lymphocytes. Protozoan leishmania, visualized as multiple gray-blue bodies within the vacuolated cytoplasm of histiocytes (hematoxylin eosin staining)

Fig. 2

Mucosal leishmaniasis. CT scan showing left maxillary sinusitis and centimetric perforation of the nasal septal cartilage