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

doi:10.12998/wjcc.v10.i16.5463

Diagnostic value of bone marrow cell morphology in visceral leishmaniasis-associated hemophagocytic syndrome: Two case reports

World J Clin Cases. 2022 Jun 6;10(16):5463-5469. doi: 10.12998/wjcc.v10.i16.5463.

Authors

Shu-Lan Shi¹, Heng Zhao², Beng-Jiang Zhou³, Ming-Biao Ma¹, Xiao-Juan Li¹, Ji Xu¹, Hong-Chao Jiang⁴

Affiliations

¹ Clinical Laboratory, The Kunming Children's Hospital, Kunming 650228, Yunnan Province, China.
² Department of Viro-immunology, Institute of Medical Biology Chinese Academy of Medical Sciences, Kunming 650118, Yunnan Province, China.
³ Department of Parasitology, Kunming Medical University Haiyuan College, Kunming 650031, Yunnan Province, China.
⁴ Yunnan Key Laboratory of Children's Major Disease Research, The Kunming Children's Hospital, Kunming 650228, Yunnan Province, China. jianghongchao@etyy.cn.

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; Case report; Hemophagocytic syndrome; Infant; Visceral leishmaniasis.

Publication types

Case Reports