Unusual Presentation of Visceral Leishmaniasis in an HIV-Infected Patient

Visceral leishmaniasis (VL) is an infectious disease caused by Leishmania infantum and is endemic in 88 countries. It is usually associated with fever, anemia, and splenomegaly, commonly defined to as “cardinal signs.” ^1,2 In areas of relatively low incidence, and with higher economic development, it can occur more frequently than expected in immune-suppressed subjects, e.g., human immunodeficiency virus (HIV)-infected transplant recipients. In these cases, the clinical presentation, diagnosis, and management may be challenging. In fact, a wide spectrum of clinical presentations has been recently reported in immunocompromised adults. ³ Lymphoid localization of Leishmania is a known, albeit rare, manifestation in immune-competent patients and is classified as “paucisyntomatic leishmaniasis” with no visceral dissemination. ⁴

Figure 1 presents a lymphonodal biopsy showing a lymphoadenopathy sustained by Leishmania.

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FIG. 1.

Lymph node. Leishmania organisms are within the cytoplasm of the macrophages. H&E 800×.

The patient, a 55-year-old man, had HIV and was receiving antiretroviral therapy with emtricitabine, tenofovir, atazanavir, and ritonavir. The treatment had been successful, achieving an undetectable HIV viral load (<50 HIV-RNA copies/ml); nevertheless, immunological discordance was observed with a modest immune recovery (CD4⁺ count always <300 cells/mm³). The patient was hospitalized because of persistent fever and leucopenia; during hospitalization splenomegaly and a generalized enlargement of the lymph nodes were detected and confirmed at CT scan, leading to a workup to investigate a possible hematological malignancy. A lymph node biopsy showed the presence of Leishmania spp. (Fig. 1). The biopsy showed a pattern characterized by Leishmania amastigotes.

In addition, Leishmania antigen was detected in urine samples and Leishmania serology (IFAT) was positive. A bone marrow biopsy revealed intracellular parasites on Giemsa staining. The patient was treated with liposomal amphotericin B and rapidly recovered. Thus, diffuse lymphoadenopathy sustained by Leishmania infantum does not always represent a benign localized disease, and may be associated with a complex clinical presentation as previously reported. ⁵ In immune-suppressed subjects with Leishmania lymphadenopathy, bone marrow and urinary antigen evaluation are indicated.

The present case was observed and evaluated within the frame of a clinical regional registry, Registro Ligure Leishmaniosi Viscerale (RiLLeVI). In areas of endemicity with a relatively low incidence, long-term registries collecting viral load cases are a useful tool to maintain a clinical perspective, to share experience concerning unusual manifestations of the disease, and to record specific local epidemiological trends.