Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jun 6, 2022; 10(16): 5463-5469
Published online Jun 6, 2022. doi: 10.12998/wjcc.v10.i16.5463
Diagnostic value of bone marrow cell morphology in visceral leishmaniasis-associated hemophagocytic syndrome: Two case reports
Shu-Lan Shi, Heng Zhao, Beng-Jiang Zhou, Ming-Biao Ma, Xiao-Juan Li, Ji Xu, Hong-Chao Jiang
Shu-Lan Shi, Ming-Biao Ma, Xiao-Juan Li, Ji Xu, Clinical Laboratory, The Kunming Children's Hospital, Kunming 650228, Yunnan Province, China
Heng Zhao, Department of Viro-immunology, Institute of Medical Biology Chinese Academy of Medical Sciences, Kunming 650118, Yunnan Province, China
Beng-Jiang Zhou, Department of Parasitology, Kunming Medical University Haiyuan College, Kunming 650031, Yunnan Province, China
Hong-Chao Jiang, Yunnan Key Laboratory of Children's Major Disease Research, The Kunming Children's Hospital, Kunming 650228, Yunnan Province, China
Author contributions: Shi SL, Zhao H, and Zhou BJ equally contributed to this manuscript; Shi SL and Zhao H wrote the manuscript and carried out the analysis; Ma MB, Li XJ, and Xu J investigated the cases; Zhou BJ and Jiang HC designed and supervised this study; and all authors read and approved the final manuscript.
Supported by the Association Foundation Program of Yunnan Science and Technology Department and Kunming Medical University, No. 2019FE001-103; Yunnan Health Training Project of High Level Talents, No. D-2017053; Top Young Experts Training Project for the Academy and Technology in Kunming and Yunnan Province, No. 202005AC160066; Postdoctoral Training Program of Yunnan Province, No. Ynbh19035; and Natural Science Foundation of Yunnan Province, No. 2019-1-C-25318000002240.
Informed consent statement: Written informed consent was obtained from the patients’ legal guardian for the publication of this case report.
Conflict-of-interest statement: The authors declare no competing interests for this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Hong-Chao Jiang, PhD, Research Scientist, Yunnan Key Laboratory of Children's Major Disease Research, The Kunming Children's Hospital, No. 288 Qianxing Road, Kunming 650228, Yunnan Province, China. jianghongchao@etyy.cn
Received: October 21, 2021
Peer-review started: October 21, 2021
First decision: February 15, 2022
Revised: March 7, 2022
Accepted: April 20, 2022
Article in press: April 20, 2022
Published online: June 6, 2022
Processing time: 224 Days and 1.7 Hours
Abstract
BACKGROUND

Visceral leishmaniasis related-hemophagocytic lymphohistiocytosis (VL-HLH) is a hemophagocytic syndrome caused by Leishmania infection. VL-HLH is rare, especially in nonendemic areas where the disease is severe, and mortality rates are high. The key to diagnosing VL-HLH is to find the pathogen; therefore, the Leishmania must be accurately identified for timely clinical treatment.

CASE SUMMARY

We retrospectively analyzed the clinical data, laboratory examination results, and bone marrow cell morphology of two children with VL-HLH diagnosed via bone marrow cell morphology at Kunming Children’s Hospital of Yunnan, China. Both cases suspected of having malignant tumors at other hospitals and who were unresponsive to treatment were transferred to Kunming Children’s Hospital. They are Han Chinese girls, one was 2 years old and the other one is 9 mo old. They had repeated fevers, pancytopenia, hepatosplenomegaly, hypertriglyceridemia, and hypofibrinogenemia over a long period and met the HLH-2004 criteria. Their HLH genetic test results were negative. Both children underwent chemotherapy as per the HLH-2004 chemotherapy regimen, but it was ineffective and accompanied by serious infections. We found Leishmania amastigotes in their bone marrow via morphological examination of their bone marrow cells, which showed hemophagocytic cells; thus, the children were diagnosed with VL-HLH. After being transferred to a specialty hospital for treatment, the condition was well-controlled.

CONCLUSION

Morphological examination of bone marrow cells plays an important role in diagnosing VL-HLH. When clinically diagnosing secondary HLH, VL-HLH should be considered in addition to common pathogens, especially in patients for whom HLH-2004 chemotherapy regimens are ineffective. For infants and young children, bone marrow cytology examinations should be performed several times and as early as possible to find the pathogens to reduce potential misdiagnoses.

Keywords: Bone marrow cell morphology; Visceral leishmaniasis; Hemophagocytic syndrome; Infant; Case report

Core Tip: This study started with the morphology of bone marrow cells, finding the pathogen from the cells, and successfully diagnosed two cases of visceral leishmaniasis related-hemophagocytic lymphohistiocytosis (VL-HLH), which was then compared and analyzed with HLH. The key criterion for differential diagnosis of VL-HLH is to find the pathogen in bone marrow cells. This has great guiding significance for clinical laboratory diagnosis and clinical treatment.