Infectious Disease

Value of metagenomic next-generation sequencing in children with visceral leishmaniasis associated with hemolytic histiocytosis

  • Lei KANG ,
  • Fang GUO ,
  • Lifang LI ,
  • Xinfeng BAI ,
  • Caiyun CHENG ,
  • Meixian XU
Expand
  • 1. Department of Pediatric Intensive Care Unit, Hebei Children’s Hospital, Shijiazhuang 050031, Hebei, China
    2. Infectious Disease Department, Hebei Children’s Hospital, Shijiazhuang 050031, Hebei, China

Received date: 2022-10-08

  Online published: 2023-08-10

Abstract

Objective To investigate the application value of metagenomic next-generation sequencing (mNGS) in pediatric visceral leishmaniasis associated with hemolytic histiocytosis (VL-HLH). Methods Clinical data of children with VL-HLH diagnosed from 1 January 2016 to 30 June 2022 were retrospectively analysed. Results A total of six VL-HLH were enrolled, three cases were imported from other province, and 3 cases were local cases. Clinical manifestations of six cases include fever, splenomegaly, pancytopenia and hyperferremia, five cases combined with hypertriglyceridemia or hypofibrinogenmia., four cases with decreased NK cell activity, three cases with increased sCD25, and two cases with phagocytosis of BMA. The VL-HLH was confirmed by BMA in two cases, and the maximum interval between onset and diagnosis were 62 and 90 days, respectively; other four cases were diagnosed by mNGS, with 34-day (16.0-39.0) interval from onset. There were five cases received glucocorticoid therapy and three cases received chemotherapy due to the delayed diagnosis, and three of them were co-infected. However, two children avoided the chemotherapy for early diagnosis by mNGS, and no co-infection occurred in them. Conclusion The clinical manifestations of VL-CMV are lack of specificity, and it is difficult to diagnose early in endemic areas of non-leishmania. mNGS can provide a basis for early diagnosis of VL-CMV and provide accurate treatment in time.

Cite this article

Lei KANG , Fang GUO , Lifang LI , Xinfeng BAI , Caiyun CHENG , Meixian XU . Value of metagenomic next-generation sequencing in children with visceral leishmaniasis associated with hemolytic histiocytosis[J]. Journal of Clinical Pediatrics, 2023 , 41(8) : 594 -598 . DOI: 10.12372/jcp.2023.22e1301

References

[1] Hines MR, von Bahr Greenwood T, Beutel G, et al. Consensus-based guidelines for the recognition, diagnosis, and management of hemophagocytic lymphohistiocytosis in critically ill children and adults[J]. Crit Care Med, 2022, 50(5): 860-872.
[2] Meeths M, Bryceson YT. Genetics and pathophysiology of haemophagocytic lymphohistiocytosis[J]. Acta Paediatr, 2021, 110(11): 2903-2911.
[3] Bolia R, Singh A, Maji M, et al. Visceral leishmaniasis associated with hemophagocytic lymphohistiocytosis[J]. Indian J Pediatr, 2021, 88(1): 73.
[4] Mantadakis E, Alexiadou S, Totikidis G, et al. A brief report and mini-review of visceral leishmaniasis-associated hemophagocytic lymphohistiocytosis in children[J]. J Pediatr Hematol Oncol, 2021, 43(2): e223-e226.
[5] Mann S, Frasca K, Scherrer S, et al. A review of leishmaniasis: current knowledge and future directions[J]. Curr Trop Med Rep, 2021, 8(2): 121-132.
[6] Henter JI, Horne A, Aricó M, et al. HLH-2004: diag-nostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis[J]. Pediatr Blood Cancer, 2007, 48(2): 124-31.
[7] Mathison BA, Bradley BT. Review of the clinical presentation, pathology, diagnosis, and treatment of leishmaniasis[J]. Lab Med, 2022, lmac134.
[8] WHO. WHO reports: leishmaniasis[EB/OL]. [2022-01-08].
[9] Guan Z, Chen C, Huang C, et al. Epidemiological features and spatial-temporal distribution of visceral leishmaniasis in mainland China: a population-based surveillance study from 2004 to 2019[J]. Parasit Vectors, 2021, 14(1): 517.
[10] Li Y, Luo Z, Hao Y, et al. Epidemiological features and spatial-temporal clustering of visceral leishmaniasis in mainland China from 2019 to 2021[J]. Front Microbiol, 2022, 13: 959901.
[11] 罗卓韦, 周正斌, 公衍峰, 等. 我国内脏利什曼病的流行现状和防控挑战[J]. 中国寄生虫学与寄生虫病杂志, 2022, 40(2): 146-152.
[12] Shi SL, Zhao H, Zhou BJ, et al. Diagnostic value of bone marrow cell morphology in visceral leishmaniasis-associated hemophagocytic syndrome: two case reports[J]. World J Clin Cases, 2022, 10(16): 5463-5469.
[13] López Marcos M, Ruiz Sáez B, Vílchez Pérez JS, et al. Distinct laboratory and clinical features of secondary hemophagocytic lymphohistiocytosis in pediatric visceral leishmaniasis: a retrospective analysis of 127 children in Andalusia, Spain (2004-2019)[J]. Pediatr Infect Dis J, 2021, 40(6): 525-530.
[14] 卫慧静, 刘小乖, 高晓鹏, 等. 儿童内脏利什曼病41例的流行病学与临床特征分析[J]. 中华传染病杂志, 2022, 40(8): 490-495.
[15] Chandrakar P, Parmar N, Descoteaux A, et al. Diffe-rential induction of SOCS isoforms by leishmania donovani impairs macrophage-T cell cross-talk and host defense[J]. Immunol, 2020, 204(3): 596-610.
[16] Bhor R, Rafati S, Pai K. Cytokine saga in visceral leishmaniasis[J]. Cytokine, 2021, 147: 155322.
[17] Ness TE, Martin-Blais R, Weatherhead JE. How I approach leishmaniasis: diagnosis and treatment in the United States[J]. J Pediatric Infect Dis Soc, 2022, 11(99): 525-532.
[18] Foinquinos J, Duarte MDC, Figueiroa JN, et al. Temporal validation of a predictive score for death in children with visceral leishmaniasis[J]. J Trop Med, 2021, 2021: 6688444.
[19] Tascini G, Lanciotti L, Sebastiani L, et al. Complex investigation of a pediatric haematological case: haemophagocytic syndrome associated with visceral leishmaniasis and epstein-barr (EBV) co-infection[J]. Int J Environ Res Public Health, 2018, 15(12): 2627.
[20] De Brito RCF, Aguiar-Soares RDO, Cardoso JMO, et al. Recent advances and new strategies in leishmaniasis diagnosis[J]. Appl Microbiol Biotechnol, 2020, 104(19): 8105-8116.
[21] Shi Q, Huang M, Li X, et al. Clinical and laboratory characteristics of hemophagocytic lymphohistiocytosis induced by Leishmania infantum infection[J]. PLoS Negl Trop Dis, 2021, 15(11): e0009944.
[22] Gao H, Wang J, Zhang S, et al. A case report of two Kala-Azar cases in China diagnosed by metagenomic next-generation sequencing[J]. Front Microbiol, 2022, 13: 922894.
[23] Graf EH, Pancholi P. Appropriate use and future directions of molecular diagnostic testing[J]. Curr Infect Dis Rep, 2020, 22(2): 5.
[24] Mottaghipisheh H, Kalantar K, Amanati A, et al. Comparison of the clinical features and outcome of children with hemophagocytic lymphohistiocytosis (HLH) secondary to visceral leishmaniasis and primary HLH: a single-center study[J]. BMC Infect Dis, 2021, 21(1): 732.
[25] Guo F, Kang L, Zhang L. mNGS for identifying pathogens in febrile neutropenic children with hematological diseases[J]. Int J Infect Dis, 2022, 116: 85-90.
Outlines

/