1. Haemophagocytic lymphohistiocytosis secondary to Plasmodium falciparum malaria: Case report and review of the literature
Fatma HAMMAMI ; Makram KOUBAA ; Rim CHAABOUNI ; Abrakhom ZEYNI ; Khaoula REKIK ; Fatma SMAOUI ; Mounir JEMAA ; Mourad CHAARI
Asian Pacific Journal of Tropical Medicine 2020;13(10):467-471
Rationale: Haemophagocytic lymphohistiocytosis is a rare complication of malaria, which is often misdiagnosed. Patient concerns: A 30-year-old male was admitted to our department for persistent fever, which began after returning from a stay in Guinea-Conakry. The laboratory investigations revealed a pancytopenia and an elevated C-reactive protein. Peripheral smear examination showed Plasmodium falciparum, therefore confirming the diagnosis of malaria. The laboratory tests showed a worsening pancytopenia. Bone marrow aspiration and biopsy revealed images of hemophagocytosis. Diagnosis: The diagnosis of haemophagocytic lymphohistiocytosis complicating malaria infection was established. Interventions: The patient was treated with artemether-lumefantrine. No immunosuppressant treatment was delivered to the patient. He received antipyretic and antimalarial treatment only. Outcomes and lessons: We report a case of haemophagocytic lymphohistiocytosis trigged by malaria infection and we review all reported cases secondary to Plasmodium falciparum malaria by searching PubMed publications till October 2019. Haemophagocytic lymphohistiocytos secondary to malaria should be suspected even in non-severe cases of malaria. Hammami Fatma 1 Infectious Diseases Department, Hedi Chaker University Hospital, University of Sfax Koubaa Makram 2 Infectious Diseases Department, Hedi Chaker University Hospital, University of Sfax Chaari Mourad 3 Hematology Laboratory, Hedi Chaker University Hospital, University of Sfax Chaabouni Rim 4 Infectious Diseases Department, Hedi Chaker University Hospital, University of Sfax Zeyni Abrakhom 5 Infectious Diseases Department, Hedi Chaker University Hospital, University of Sfax Rekik Khaoula 6 Infectious Diseases Department, Hedi Chaker University Hospital, University of Sfax Smaoui Fatma 7 Infectious Diseases Department, Hedi Chaker University Hospital, University of Sfax Jemaa Mounir 8 Infectious Diseases Department, Hedi Chaker University Hospital, University of Sfax Nascimento TL do, Vasconcelos SP, Peres Y, Oliveira MJS de, Taminato M, Souza KMJ de. Prevalence of malaria relapse: Systematic review with meta-analysis. Rev Lat Am Enfermagem 2019; 18(27): e3111. Talapko J, Skrlec I, Alebic T, Jukic M, Vcev A. Malaria: The past and the present. Microorganisms 2019; 7(6): 179. Gzara Zargouni A, Tej Dellagi R, Ben Alaya N, Ben Jemaa N, Gamara D, Ben Salah A, et al. Indigenous malaria in Tunisia: 4 cases registered in 2013 in Tunisia. Tunis Med 2015; 93(8-9): 543-547. Jiménez-Morillas F, Gil-Mosquera M, García-Lamberechts EJ. Fever in travellers returning from the tropics. Med Clin (Barc) 2019; 153(5): 205-212. Amireh S, Shaaban H, Guron G. Severe Plasmodium vivax cerebral malaria complicated by hemophagocytic lymphohistiocytosis treated with artesunate and doxycycline. Hematol Oncol Stem Cell Ther 2018; 11(1): 34-37. Muthu V, Dhooria S, Sehgal IS, Agarwal R, Behera D, Varma N. Malaria-associated secondary haemophagocytic lymphohistiocytosis: Report of two cases & a review of literature. Indian J Med Res 2017; 145(3): 399-404. Jordan MB, Allen CE, Greenberg J, Henry M, Hermiston ML, Kumar A, et al. Challenges in the diagnosis of hemophagocytic lymphohistiocytosis: Recommendations from the North American Consortium for Histiocytosis (NACHO). Pediatr Blood Cancer 2019; 66(11): e27929. Harioly Nirina MOM, Raheritiana TM, Harioly Nirina MOJ, Rasolonjatovo AS, Rakoto Alson AO, Rasamindrakotroka A. Hemophagocytic lymphohistiocytosis associated with Plasmodium falciparum. Med Mal Infect 2017; 47(8): 569-570. Niang A, Niang SE, Ka EHF, Ka MM, Diouf B. Collapsing glomerulopathy and haemophagocytic syndrome related to malaria: A case report. Nephrol Dial Transplant 2008; 23(10): 3359-3361. Ohnishi K, Mitsui K, Komiya N, Iwasaki N, Akashi A, Hamabe Y. Clinical case report: Falciparum malaria with hemophagocytic syndrome. Am J Trop Med Hyg 2007; 76(6): 1016-1018. Ladeb S, Ben Abdejlil N, Fakhfakh N, Lakhal A, Belloumi D, Ben Hamed L, et al. Plasmodium falciparum infection transmitted by transfusion: A cause of hemophagocytic syndrome after bone marrow tranplantation in a non-endemic country. Transpl Infect Dis 2018; 20(3): e12887. Selvarajan D, Sundaravel S, Alagusundaramoorthy SS, Jacob A. Hemophagocytic lymphohistiocytosis with concurrent malarial infection. BMJ Case Rep 2017; 2017: bcr2017221159. Weeratunga P, Rathnayake G, Sivashangar A, Karunanayake P, Gnanathasan A, Chang T. Plasmodium falciparum and Mycoplasma pneumoniae co-infection presenting with cerebral malaria manifesting orofacial dyskinesia and haemophagocytic lymphohistiocytosis. Malar J 2016; 15: 461. Bhagat M, Kanhere S, Kadakia P, Phadke V, George R, Chaudhari K. Haemophagocytic lymphohistiocytosis: A cause of unresponsive malaria in a 5-year-old girl. Paediatr Int Child Health 2015; 35(4): 333-336. Trifi A, Terra A, Abdellatif S, Daly F, Oueslati M, Laamari L, et al. Haemophagocytic lymphohistiocytosis syndrome-related to severe malaria: rare association. Tunis Med 2014; 92(7): 515-516. Santos JA, Neves JF, Venâncio P, Gouveia C, Varandas L. Hemophagocytic lymphohistiocytosis secondary to Falciparum malaria in a 5 year-old boy. Ann Hematol 2015; 94(1): 161-163. Vinoth PN, Thomas KA, Selvan SM, Suman DFR, Scott JX. Hemophagocytic syndrome associated with Plasmodium falciparum infection. Indian J Pathol Microbiol 2011; 54(3): 594-596. Dass R, Barman H, Duwarah SG, Choudhury V, Jain P, Deka NM, et al. Macrophage activation syndrome in malaria. Rheumatol Int 2010; 30(8): 1099-1101. Sermet-Gaudelus I, Abadie V, Stambouli F, Hennequin C, Lenoir G, Gendrel D. Haemophagocytic syndrome in Plasmodium falciparum malaria. Acta Paediatr 2000; 89(3): 368-369. Zvulunov A, Tamary H, Gal N. Pancytopenia resulting from hemophagocytosis in malaria. Pediatr Infect Dis J 2002; 21(11): 1086-1087. Sanklecha M, Mehta N, Bagban H. Varied presentation of complicated falciparum malaria in a family. Indian Pediatr 2012; 49(5): 413-414. Abdelkefi A, Ben Othman T, Torjman L, Ladeb S, Lakhal A, Belhadj S, et al. Plasmodium falciparum causing hemophagocytic syndrome after allogeneic blood stem cell transplantation. Hematol J Off J Eur Haematol Assoc 2004; 5(5): 449-450. Rehman JU, Bhabri N, Waleed A, Maulawi A, Aslam M. Falciparum malaria in a patient with sickle cell trait with hemophagocytosis and secondary pancytopenia. Ann Hematol 2012; 91(8): 1329-1330. Retornaz F, Seux V, Arnoulet C, Durand JM, Sainty D, Soubeyrand J. Plasmodium falciparum malaria infection complicated by haemophagocytic syndrome in an old man. Acta Haematol 2000; 103(4): 224-225. Anwar M, Saleem M, Malik IA. Severe haemophagocytic syndrome in falciparum malaria. JPak Med Assoc 1995; 45(11): 302-303. Ohno T, Shirasaka A, Sugiyama T, Furukawa H. Hemophagocytic syndrome induced by Plasmodium falciparum malaria infection. Int J Hematol 1996; 64(3-4): 263-266. Epelboin L, Rapp C, Faucher JF, Mechai' F, Bottieau E, Matheron S, et al. Management and treatment of uncomplicated imported malaria in adults. Update of the French malaria clinical guidelines. Med Mal Infect 2020; 50(2):194-212. Tothova Z, Berliner N. Hemophagocytic syndrome and critical illness: New insights into diagnosis and management. J Intensive Care Med 2015; 30(7): 401-412. Malinowska I, Machaczka M, Popko K, Siwicka A, Salamonowicz M, Nasilowska-Adamska B. Hemophagocytic syndrome in children and adults. Arch Immunol Ther Exp (Warsz) 2014; 62(5): 385-394.