Hemophagocytic lymphohistiocytosis secondary to visceral leishmaniasis in an endemic area in the north of Minas Gerais, Brazil

Carvalho, Fernando Henrique Guimarães de; Lula, Jamille Fernandes; Teles, Leandro de Freitas; Caldeira, Antônio Prates; Carvalho, Sílvio Fernando Guimarães

doi:10.1590/0037-8682-0491-2019

Cited by 11 publications

(8 citation statements)

References 31 publications

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“…Chemotherapy including methylprednisolone, cyclosporine A and etoposide is recommended particularly due to corticosteroids as the important components [ 15 , 32 , 33 ]. Indeed, the combination of anti- Leishmania and anti- inflammatory therapy facilitated the remission in the VL associated HLH patients [ 34 ].…”

Section: Discussionmentioning

confidence: 99%

Clinical and laboratory characteristics of hemophagocytic lymphohistiocytosis induced by Leishmania infantum infection

Shi

Huang

et al. 2021

PLoS Negl Trop Dis

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Background Visceral leishmaniasis (VL) could progress to secondary hemophagocytic lymphohistiocytosis (HLH), which is a rare but life-threatening condition with poor prognosis. So far, the clinical and laboratory characteristics of VL associated HLH have not been well elucidated. Method and findings In this study, we retrospectively analyzed the clinical and laboratory profiles between 17 patients with VL associated HLH and 27 patients with VL alone admitted at the Beijing Friendship Hospital, Capital Medical University from May 2016 to March 2021. In addition to the identification of Leishmania infection, hemophagocytosis was identified in bone marrow in the most cases of VL associated HLH (15/17). The patients with VL associated HLH had higher chances of bleeding, hepatomegaly, thrombocytopenia, hypertriglyceridemia, hyperferritinemia, hypofibrinogenemia, elevated secretion of soluble IL-2 receptor or lower NK cell activity compared to patients with VL only. Furthermore, patients with VL associated HLH had higher inflammation status associated with higher levels of Th1 (TNF-α, IFN-γ, IL-1beta, IL-6, IL-8, IL-12p70), Th2 (IL-4) and Th17 cytokines (IL-17, IL-23) in the peripheral blood, and higher parasite load (qPCR and parasite culture). All 27 VL cases were totally recovered after being treated with Sodium Stibogluconate, five of the 17 patients with VL associated HLH died even after timely treatment with anti-parasite and immunosuppressive chemotherapy. Conclusion Without appropriate treatment, visceral leishmaniosis could develop to secondary HLH. The parasite culturing and qPCR detection of bone marrow samples facilitates the diagnosis of VL associated HLH in addition to other findings of HLH. Prompt treatment with anti-Leishmania and immunosuppressive chemotherapy is critical to reduce the mortality of VL associated HLH.

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Section: Discussionmentioning

confidence: 99%

Clinical and laboratory characteristics of hemophagocytic lymphohistiocytosis induced by Leishmania infantum infection

Shi

Huang

et al. 2021

PLoS Negl Trop Dis

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“…This syndrome is frequent in children, and it is a common circumstance of VL in this population [7]. In adults, VL has various symptoms, but HLH is rare [7]. In our case, VL was discovered unexpectedly after bone marrow investigation due to confirmation of HLH diagnosis.…”

Section: Case Descriptionmentioning

confidence: 63%

“…Pancytopenia, elevated levels of serum ferritin and triglycerides, coagulopathy with hypofibrinogenemia, and abnormal liver enzymes are commonly present. This syndrome is frequent in children, and it is a common circumstance of VL in this population [7]. In adults, VL has various symptoms, but HLH is rare [7].…”

Section: Case Descriptionmentioning

confidence: 94%

Unusual occurrence of hemophagocytic lymphohistiocytosis in HIV-positive person with visceral leishmaniasis

Kooli¹,

Marrakchi²,

Toumi³

et al. 2021

HIV & AIDS Review

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Introduction:Visceral leishmaniasis is a well-recognized opportunistic infection in people living with HIV (PLHIV). Unlike adults, in children this infection is frequently associated with hemophagocytic lymphohistiocytosis (HLH). We report a case of HLH in HIV-positive person with visceral leishmaniasis.Case description: A 25-year-old man known living with HIV since 2013 was admitted to infectious diseases department in March 2017. His clinical examination was clear. His initial viral load was 630,000 copies/mm 3 and CD4+ cells count was 12/mm 3 . No opportunistic infections were noted. The patient was started on antiretroviral therapy. During hospitalization, he developed fever, asthenia, rhinorrhea, and odynophagia. The diagnosis of HLH was retained because of pancytopenia, cytolysis, hyponatremia, high level of ferrinemia, and hemophagocytosis. Etiological investigations revealed positive Leishmania PCR. Also, Leishmania was detected in sternal puncture. Patient received meglumine antimoniate (glucantime) 20 mg/kg/day for 21 days with favorable outcomes. To prevent relapse, he received meglumine antimoniate 20 mg/kg/month as long as his CD4+ count was less than 100 cells/mm 3 . After 1-year follow up, no relapse was detected.Conclusions: Clinical and laboratory presentation of visceral leishmaniasis in PLHIV may differ from classic kala-azar. In our case, HLH was the reason for VL discovery.

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“…Em relação à terapia específica para LHH, não houve necessidade de sua implementação, visto que a paciente apresentou melhora clínica e remissão do quadro com uso de desoxicolato de anfotericina B para a causa-base. pacientes com LHH secundária a LV tratados para a doença de base obtiveram sucesso terapêutico, assim como apontado por Carvalho et al (2020). Sendo assim, embora a resolução da LV não garanta a melhora da LHH secundária a ela, há diversos relatos na literatura médica de casos similares ao relatado que evoluíram favoravelmente para remissão do quadro de LHH pelo manejo da infecção pelo Leishmania.…”

Section: Discussionunclassified

“…Diante desse cenário, o uso da corticoterapia ainda é duvidosa, sendo necessárias mais evidências para indicar a melhor opção. A anfotericina B lipossomal é eficiente e bem tolerada quando utilizada por curto período, porém há critérios para a sua utilização em casos de LV (CARVALHO et al, 2020).…”

Section: Em Geral é Fundamental Que O Tratamento Seja Precoce E Agres...unclassified

Síndrome hemofagocítica pós-leishmaniose visceral: revisão de literatura e relato de caso

Souza¹,

Pizzol²,

Leite³

et al. 2022

Braz. J. Hea. Rev.

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Introdução: A Síndrome Hemofagocítica (SH), também chamada de Linfo-histiocitose Hemofagocítica (LHH), é uma doença rara, de acometimento multissistêmico resultante da ativação excessiva de linfócitos T citotóxicos, células natural killer e macrófagos, levando à secreção de citocinas pró-inflamatórias. Quando associada à Leishmaniose Visceral (LV) se torna um quadro de difícil diagnóstico, visto que os sintomas são semelhantes. Objetivos: Realizar relato do caso e revisão bibliográfica sobre HHpós-LV em uma paciente pediátrica. Relato do caso: Paciente 1 ano e 8 meses, apresentava história de febre iniciada há cerca de 15 dias, evoluindo com prostração, hiporexia, distensão abdominal, hepatoesplenomegalia e palidez. À admissão hospitalar, apresentou resultado de teste rápido positivo para LV, e recebeu tratamento com desoxicolato de anfotericina B, 1mg/kg/dia, em uma dose diária. Posteriormente, o diagnóstico foi confirmado pelo exame de PCR. A paciente recebeu alta hospitalar após 22 dias de internação. Discussão: O uso de anfotericina B lipossomal leva à remissão permanente do quadro, entretanto nem sempre a terapia antiparasitária será resolutiva para a LHH/LV e, devido à alta letalidade da doença, há a necessidade de iniciar medicamentos imunossupressores ou antiinflamatórios em caráter imediato para evitar um desfecho fatal. No caso clínico discutido, optou-se por iniciar anfotericina B para tratamento da LV, com desfecho positivo para a paciente. A paciente não precisou iniciar o esquema terapêutico imunossupressor, uma vez que a monoterapia antiparasitária foi resolutiva. Evoluiu favoravelmente, recebendo alta para domicílio no 22º dia de internação. Conclusão: A LHH deve ser considerada suspeita em casos de LV de evolução lenta, com febre prolongada, esplenomegalia e citopenias refratárias, especialmente em crianças de área endêmica, considerando que é uma condição potencialmente fatal se não tratada. Vale enfatizar a semelhança clínica e do hemograma em ambas as doenças, o que torna o diagnóstico complexo e, consequentemente, atrasa o tratamento.

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Hemophagocytic lymphohistiocytosis secondary to visceral leishmaniasis in an endemic area in the north of Minas Gerais, Brazil

Cited by 11 publications

References 31 publications

Clinical and laboratory characteristics of hemophagocytic lymphohistiocytosis induced by Leishmania infantum infection

Clinical and laboratory characteristics of hemophagocytic lymphohistiocytosis induced by Leishmania infantum infection

Unusual occurrence of hemophagocytic lymphohistiocytosis in HIV-positive person with visceral leishmaniasis

Síndrome hemofagocítica pós-leishmaniose visceral: revisão de literatura e relato de caso

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