Atypical clinical presentation of visceral leishmaniasis: a rare case without splenomegaly

Case report

A 59-year-old woman, resident in an endemic area for visceral leishmaniasis (VL), was admitted to our hospital (a referral centre for infectious disease) with fever, anorexia and weakness for the previous three months. Over the previous three days, she had gingival bleeding, lumbar pain and macroscopic haematuria. Physical examination revealed a pyrexia of 38.4℃ and significant pallor. There was no hepatosplenomegaly nor palpable lymph nodes; the remaining examination was unremarkable. Laboratorial examination revealed pancytopenia and an inversion of the normal albumin/globulin ratio. HIV serology was negative. Abdominal computed tomography (CT) showed no evidence of organomegaly. The diagnosis of VL was established by the demonstration of Leishmania amastigotes in bone marrow smears stained with Giemsa (Figure 1). ELISA using recombinant K39 antigen (anti-rK39 IgG) was also positive, but a rapid immunochromatographic test (ICT) using the same antigen (Kalazar detect®) was negative. Therapy with amphotericin B was initiated for seven days, with a good clinical and laboratory response: complete resolution of pancytopenia, fever and bleeding, including her haematuria. Although schistosomiasis is endemic in Brazil it does not normally affect the urinary tract, so no specific further urinary testing was carried out. Because of her excellent recovery, no control bone marrow aspiration screen was deemed necessary. She remained asymptomatic at follow up. OPEN IN VIEWER

In fully established VL, fever, weakness, weight loss and hepatosplenomegaly are common features. Splenomegaly is the hallmark of the disease and is frequently more prominent than hepatomegaly. ¹ In our case neither was present, which is rare.^2–4 In immunocompetent individuals, splenomegaly is described in up to 98% of cases. The absence of organomegaly is more commonly reported in immunosuppressed patients. ⁵ Our patient showed no clinical sign of immune compromise, although it is possible that a hidden pathology may have been present. Nonetheless, the possibility of LV without hepatosplenomegaly must still be considered in an endemic area.