Abstract:Visceral leishmaniasis (VL) is a neglected tropical disease with more than 30,000 cases annually reported worldwide. In Brazil, about 3,700 cases are annually reported. The VL clinical presentation is variable, from asymptomatic to severe cases with a high risk of death. We reported three cases of VL with clinical sign similarities but distinct development. All cases had bone marrow hemophagocytosis and hemophagocytic lymphohistiocytosis (HLH) criteria. HLH is a rare condition that may have secondary causes, i… Show more
“…None of the inclusions resembled H. capsulatum yeast forms, and were thought to represent phagocytized cellular and/or some form of lipid debris. Despite the microscopic conclusions, however, liposomal amphotericin B (10 mg/kg on 2 consecutive days [7] ) was nevertheless given for the remote possibility of Mediterranean visceral leishmaniasis associated with HLH [1] , [2] , [3] . Urgent treatment by the medical staff was prompted out of an abundance of clinical caution -- at the time of bone marrow examination, the patient was febrile, rapidly deteriorating and requiring intensive care unit support.…”
Section: Discussionmentioning
confidence: 99%
“…1 ). This finding, along with travel in the previous 6 months in both southern Europe and the Caribbean region, raised the question of visceral leishmaniasis (kala-azar) as well as disseminated histoplasmosis, both of which have been associated with secondary HLH [1] , [2] , [3] , [4] . Fig.…”
“…None of the inclusions resembled H. capsulatum yeast forms, and were thought to represent phagocytized cellular and/or some form of lipid debris. Despite the microscopic conclusions, however, liposomal amphotericin B (10 mg/kg on 2 consecutive days [7] ) was nevertheless given for the remote possibility of Mediterranean visceral leishmaniasis associated with HLH [1] , [2] , [3] . Urgent treatment by the medical staff was prompted out of an abundance of clinical caution -- at the time of bone marrow examination, the patient was febrile, rapidly deteriorating and requiring intensive care unit support.…”
Section: Discussionmentioning
confidence: 99%
“…1 ). This finding, along with travel in the previous 6 months in both southern Europe and the Caribbean region, raised the question of visceral leishmaniasis (kala-azar) as well as disseminated histoplasmosis, both of which have been associated with secondary HLH [1] , [2] , [3] , [4] . Fig.…”
Hemophagocytic lymphohistiocytosis (HLH) is a rare and fatal complication of visceral leishmaniasis (VL). To provide a basis for early and correct diagnosis and to improve prognosis in the future, we describe a case series of VL-associated HLH in adults in our center in the past decade after review of all reported cases of adult VL-associated HLH in English through May 2022. In our case series, a total of 111 patients were diagnosed with VL. Among these patients, only six cases were diagnosed with VL-associated HLH. All patients tested positive for serology. Leishmania was detected for the first time by bone marrow aspiration (BMA) in three of the six patients and in the other three patients after three or four BMAs. It took more than 1 month from onset to diagnosis of VL for all the six cases, and the longest time was 6 months. Five of the six patients recovered after receiving sodium stibogluconate. VL-associated HLH is rare but potentially life-threatening in adults and predisposes to early delays in diagnosis. However, diagnostic techniques are not complicated or difficult, so it is more important to consider that it is not recognized by physicians. Although guidelines recommend liposomal amphotericin B as the most effective therapy, our experience suggests that sodium stibogluconate can be an alternative option when liposomal amphotericin B is unavailable or unaffordable.
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