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2020, Number 3

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Rev Cubana Med Trop 2020; 72 (3)

Hemophagocytic lymphohistiocytosis associated with visceral leishmaniasis. Review of cases reported

Garrido D, Fuseau M, Garrido S, Celi S

Language: English
References: 40
Page: 1-16
PDF size: 459.97 Kb.


ABSTRACT

Introduction: Leishmaniasis is a tropical and subtropical disease highly reported in Southeast Asia, East Africa, Latin America, and the Mediterranean basin, with an incidence of two million new cases by year and 500,000 cases of visceral leishmaniasis. One of the more severe and rare complications of visceral leishmaniasis is hemophagocytic lymphohistiocytosis.
Objective: To describe the clinical characteristics of hemophagocytic lymphohistiocytosis associated with visceral leishmaniasis.
Methods: We performed a literature review based on the case reports indexed in MEDLINE/PubMed.
Results: Twenty-five cases were included; 52 % under two years of age. All cases presented splenomegaly and 84% hepatomegaly. Cytopenias were described in all patients: 100% thrombocytopenia, 96% anemia, and 84% leukopenia or neutropenia. Hypertriglyceridemia and hypofibrinogenemia were found in 68% and 32%, respectively, and hyperferritinemia in 80%. Additionally, hemophagocytosis was documented in 84%, with Leishmania detection in 92%. All patients were treated against Leishmania: 80% with liposomal amphotericin B. regarding the treatment for hemophagocytic lymphohistiocytosis; corticosteroid were used in 36%, endovenous immunoglobulin in 28%, cyclosporine in 28% and etoposide in 16% The complications reported included gastrointestinal hemorrhage (8%), disseminated intravascular coagulation (8%), autoimmune hemolytic anemia (12%), multiple-organ dysfunction/septic shock (12%), petechial rash (16%), and four patients deceased. Variables such as fever (p=0.031), hemoglobin level (p=0.031), platelet count (p=0.0048), and ferritin (p=0.0072) were associated with mortality.
Conclusions: During visceral leishmaniasis, the hemophagocytic syndrome is a rare condition that mainly affects pediatric patients, but with excellent outcomes using liposomal amphotericin B. However, there is a lack of strong evidence to make a recommendation.


REFERENCES

  1. Burza S, Croft SL, Boelaert M. Leishmaniasis. Lancet. 2018;392(10151):951-70

  2. Torres-Guerrero E, Quintanilla-Cedillo MR, Ruiz-Esmenjaud J, Arenas R. Leishmaniasis: a review. F1000Res 2017;6:750. doi:10.12688/f1000research.11120.1

  3. World Health Organization. Leishmaniasis; Epidemiological situation [Internet] 2018. [access: 14/03/20]. Available from: Available from: https://www.who.int/leishmaniasis/burden/en/

  4. Jamka GE, Lehmberg K. Hemophagocytic lymphohistiocytosis: pathogenesis and treatment. Hematology Am Soc Hematol Educ Program. 2013;2013:605-11. doi: 10.1182/asheducation-2013.1.605

  5. George MR. Hemophagocytic lymphohistiocytosis: review of etiologies and management. J Blood Med. 2014;5:69-86. doi:10.2147/JBM.S46255

  6. Morimoto A, Omachi S, Osada Y, Chambers JK, Uchida K, Sanjoba C, et al. Hemophagocytosis in Experimental Visceral Leishmaniasis by Leishmania donovani. PLoS Negl Trop Dis. 2016;10(3):e0004505. doi:10.1371/journal.pntd.0004505

  7. George JT, Sadiq M, Sigamani E, Mathuram AJ. Visceral leishmaniasis with haemophagocytic lymphohistiocytosis. BMJ Case Rep. 2019;12(2):e226361. doi:10.1136/bcr-2018-226361

  8. Tascini G, Lanciotti L, Sebastiani L, Paglino A, Esposito S. Complex Investigation of a Pediatric Haematological Case: Haemophagocytic Syndrome Associated with Visceral Leishmaniasis and Epstein?Barr (EBV) Co-Infection. Int J Environ Res Public Health. 2018;15(12):e2672. doi: 10.3390/ijerph15122672

  9. Costa A, Pais C, Cerqueira S, Salvador F. Visceral Leishmaniasis Associated with Macrophage Activation Syndrome and Diffuse Alveolar Hemorrhage in a Lupus Patient. Acta Med Port. 2018;31(10):593-6.

  10. Adamczick C, Dierig A, Welzel T, Schifferli A, Blum J, Ritz N. Double trouble: visceral leishmaniasis in twins after traveling to Tuscany - a case report. BMC Infect Dis. 2018;18(1):495.

  11. García Pérez J, Recio Iglesias J, Olivé Gadea M. Visceral leishmania-associated hemophagocytic lymphohistiocytosis. Med Clin (Barc). 2018;151(5):e25-e26.

  12. Gaifer Z, Boulassel MR. Leishmania Infantum and Epstein-Barr Virus Co-Infection in a Patient with Hemophagocytosis. Infect Dis Rep. 2016;8(4):6545. doi:10.4081/idr.2016.6545

  13. Aydin Teke T, Metin Timur Ö, Gayretli Aydin ZG, Öz N, Bayhan GI, Yilmaz N, et al. Three Pediatric Cases of Leishmaniasis with Different Clinical Forms and Treatment Regimens. Turkiye Parazitol Derg. 2015;39(2):147-50

  14. Rios-Fernández R, Callejas-Rubio JL, García-Rodríguez S, Sancho J, Zubiaur M, Ortego-Centeno N. Tocilizumab as an Adjuvant Therapy for Hemophagocytic Lymphohistiocytosis Associated With Visceral Leishmaniasis. Am J Ther. 2016;23(5):e1193-6.

  15. Higel L, Froehlich C, Pages MP, Dupont D, Collardeau-Frachon S, Dijoud F, et al. Macrophage activation syndrome and autoimmunity due to visceral leishmaniasis. Arch Pediatr. 2015;22(4):397-400.

  16. DomInguez-Pinilla N, Baro-Fernández M, González-Granado LI. Hemophagocytic lymphohistiocytosis secondary to Epstein Barr virus and Leishmania co-infection in a toddler. J Postgrad Med. 2015;61(1):44-5. doi:10.4103/0022-3859.147052

  17. Watkins ER, Shamasunder S, Cascino T, White KL, Katrak S, Bern C, et al. Visceral leishmaniasis-associated hemophagocytic lymphohistiocytosis in a traveler returning from a pilgrimage to the Camino de Santiago. J Travel Med. 2014;21(6):429-32.

  18. Visentin S, Baudesson de Chanville A, Loosveld M, Chambost H, Barlogis V. Infantile visceral leishmaniasis, an etiology of easily curable hemophagocytic lymphohistiocytosis syndrome. Arch Pediatr. 2013;20(11):1225-9.

  19. Singh G, Shabani-Rad MT, Vanderkooi OG, Vayalumkal JV, Kuhn SM, Guilcher GM, et al. Leishmania in HLH: a rare finding with significant treatment implications. J Pediatr Hematol Oncol. 2013;35(3):e127-9.

  20. Ay Y, Yildiz B, Unver H, Karapinar DY, Vardar F. Hemophagocytic lymphohistiocytosis associated with H1N1 virus infection and visceral leishmaniasis in a 4.5-month-old infant. Rev Soc Bras Med Trop. 2012;45(3):407-9.

  21. Bouguila J, Chabchoub I, Moncef Y, Mlika A, Saghrouni F, Boughamoura L, et al. Treatment of severe hemophagocytic syndrome associated with visceral leishmaniasis. Arch Pediatr 2010;17(11):1566-70.

  22. Levy L, Nasereddin A, Rav-Acha M, Kedmi M, Rund D, Gatt ME. Prolonged fever, hepatosplenomegaly, and pancytopenia in a 46-year-old woman. PLoS Med. 2009;6(4):e1000053. doi:10.1371/journal.pmed.1000053

  23. Koliou MG, Soteriades ES, Ephros M, Mazeris A, Antoniou M, Elia A, et al. Hemophagocytic lymphohistiocytosis associated with Epstein Barr virus and Leishmania donovani coinfection in a child from Cyprus. J Pediatr Hematol Oncol. 2008;30(9):704-7.

  24. Celik U, Alabaz D, Alhan E, Bayram I, Celik T. Diagnostic dilemma in an adolescent boy: hemophagocytic syndrome in association with kala azar. Am J Med Sci. 2007;334(2):139-41.

  25. Tapisiz A, Belet N, Ciftçi E, Ince E, Dogru U. Hemophagocytic lymphohistiocytosis associated with visceral leishmaniasis. J Trop Pediatr. 2007;53(5):359-61

  26. Kilani B, Ammari L, Kanoun F, Ben Chaabane T, Abdellatif S, Chaker E. Hemophagocytic syndrome associated with visceral leishmaniasis.Int J Infect Dis. 2006;10(1):85-6.

  27. Ozyürek E, Ozçay F, Yilmaz B, Ozbek N. Hemophagocytic lymphohistiocytosis associated with visceral leishmaniasis: a case report. Pediatr Hematol Oncol. 2005;22(5):409-14.

  28. Marom D, Offer I, Tamary H, Jaffe CL, Garty BZ. Hemophagocytic lymphohistiocytosis associated with visceral leishmaniasis. Pediatr Hematol Oncol. 2001;18(1):65-70.

  29. Parikh SA, Kapoor P, Letendre L, Kumar S, Wolanskyj AP. Prognostic factors and outcomes of adults with hemophagocytic lymphohistiocytosis. Mayo Clin Proc. 2014;89(4):484-92.

  30. Rivière S, Galicier L, Coppo P, Marzac C, Aumont C, Lambotte O, et al. Reactive hemophagocytic syndrome in adults: a retrospective analysis of 162 patients. Am J Med. 2014;127(11):1118-25.

  31. Grzybowski B, Vishwanath VA. Hemophagocytic Lymphohistiocytosis: A Diagnostic Conundrum. J Pediatr Neurosci. 2017;12(1):55-60. doi:10.4103/jpn.JPN_140_16

  32. George MR. Hemophagocytic lymphohistiocytosis: review of etiologies and management. J Blood Med. 2014;5:69-86. doi:10.2147/JBM.S46255

  33. Shu MM, Zhu HF, Zhang T, Gao GX, Chen XQ, et al. Clinical analysis on 28 patients with he1ophagocytic lymphohistiocytosis syndrome. Zhongguo Shi Yan Xue Ye Xue Za Zhi. 2010;18(2):463-5.

  34. Rajagopala S, Singh N. Diagnosing and treating hemophagocytic lymphohistiocytosis in the tropics: systematic review from the Indian subcontinent. Acta Med Acad. 2012;41(2):161-74.

  35. Bode SF, Bogdan C, Beutel K, Behnisch W, Greiner J, et al. Hemophagocytic lymphohistiocytosis in imported pediatric visceral leishmaniasis in a nonendemic area. J Pediatr. 2014;165(1):147-53.

  36. Aronson N, Herwaldt BL, Libman M. Diagnosis and Treatment of Leishmaniasis: Clinical Practice Guidelines by the Infectious Diseases Society of America (IDSA) and the American Society of Tropical Medicine and Hygiene (ASTMH). Am J Trop Med Hyg. 2017;96(1):24-45. doi:10.4269/ajtmh.16-84256

  37. Jordan MB, Allen CE, Weitzman S, Filipovich AH, McClain KL. How I treat hemophagocytic lymphohistiocytosis. Blood. 2011;118(15):4041-52. doi:10.1182/blood-2011-03-278127

  38. Sampaio MJ, Cavalcanti NV, Alves JG, Filho MJ, Correia JB. Risk factors for death in children with visceral leishmaniasis. PLoS Negl Trop Dis. 2010;4(11):e877. doi:10.1371/journal.pntd.0000877

  39. Ben Helel K, Ben Rejeb M, Habboul Z, Khattat N, Mejaouel H, Said-Latiri H, et al. Risk factors for mortality of children with zoonotic visceral leishmaniasis in Central Tunisia. PLoS One. 2017;12(12):e0189725. doi:10.1371/journal.pone.0189725

  40. Druzian AF, de Souza AS, de Campos DN, Croda J, Higa MG, Dorval ME, et al. Risk Factors for Death from Visceral Leishmaniasis in an Urban Area of Brazil. PLoS Negl Trop Dis 2015;9(8):e0003982. doi:10.1371/journal.pntd.0003982




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