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CASE REPORT article
Front. Pediatr.
Sec. Pediatric Infectious Diseases
Volume 13 - 2025 | doi: 10.3389/fped.2025.1561600
Hemophagocytic Lymphohistiocytosis Secondary to Visceral Leishmaniasis in Children: Case Report and Systematic Review
Provisionally accepted- 1 Department of Pediatric Cardiology, Chengdu Women and Children’s Central Hospital, Chengdu, Sichuan Province, China
- 2 School of Medicine, University of Electronic Science and Technology of China, Chengdu, Sichuan Province, China
- 3 Department of Pediatrics, Public Health and Clinical Center of Chengdu, Chengdu, China
- 4 Department of Pediatrics, First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou, Zhejiang Province, China
Background: Visceral leishmaniasis (VL) can lead to complications such as hemophagocytic lymphohistiocytosis (HLH) in children. The clinical features of VL overlap with that of HLH, and thus the diagnosis of VL-induced HLH can be challenging for clinicians.Methods: We describe two pediatric cases and systematically review all reported cases of pediatric VL-related HLH in literatures until May 2024.Results: The demographic characteristics, clinical manifestations, treatment and prognosis of our reported cases are presented. The systematic review included 29 articles with a total of 135 cases. More than half of the children (89/125, 71.2%) were under 3 years old, and 8.9% (n=12/135) had specific epidemiological histories. The main clinical presentations were hypertriglyceridemia (34/45, 75.6%), hypofibrinogenemia (24/36, 66.7%), and hyperferritinemia (95/132, 72.0%). Bone marrow aspiration (BMA) analysis indicated positive evidence of leishmania infection in 84.7% (83/98) of cases, while 37.8% (14/37) of patients tested negative for leishmania on the first BMA smear. All patients were treated against leishmania with amphotericin B (76/135, 56.3%) or antimony (77/135, 57.0%), and 13.3% (n=18/135) of patients received both medications, in which amphotericin B was used as rescue treatment. The prognosis was favorable, with the exception of two deaths.Conclusions: Vigilance towards screening for leishmania infection induced HLH is imperative, particularly when there is a suspicious epidemiological history, ineffective chemotherapy, or prior to bone marrow transplantation. Early recognition, accurate diagnosis, and prompt treatment initiation can significantly alter the course of the disease and favor the prognosis in childhood with HLH secondary to VL.
Keywords: Visceral leishmaniasis, hemophagocytic lymphohistiocytosis, Children, Amphotericin B, Systematic review
Received: 16 Jan 2025; Accepted: 24 Mar 2025.
Copyright: © 2025 Chen, Gao, Zhang and Mao. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
* Correspondence:
Yi Gao, Department of Pediatrics, Public Health and Clinical Center of Chengdu, Chengdu, China
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