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Paroxysmal cold hemoglobinuria is a rare autoimmune hemolytic anemia seen almost exclusively in children under 5 years of age after a viral illness. It is mediated by a biphasic polyclonal autoantibody against red blood cells, which causes severe hemolysis that typically self-resolves within 2 weeks without recurrence. While laboratory identification of the aforementioned antibody, the Donath Landsteiner antibody, would confirm this diagnosis, a negative test does not rule out this condition in the appropriate clinical context. We report on the rare occurrence of a severe presentation of paroxysmal cold hemoglobinuria in a 17-year-old male with Epstein-Barr virus infection.

Competing Interests: The authors have no conflicts of interest to declare.

Titel:	Epstein-Barr Virus-induced Paroxysmal Cold Hemoglobinuria in a 17-Year-old Male.
Autor/in / Beteiligte Person:	Schmittau, KM ; Giordano, L
Zeitschrift:	Journal of pediatric hematology/oncology, Jg. 45 (2023-11-01), Heft 8, S. e1014-e1017
Veröffentlichung:	1998- : Hagerstown, MD : Lippincott Williams & Wilkins ; <i>Original Publication</i>: New York, NY : Raven Press, c1995-, 2023
Medientyp:	academicJournal
ISSN:	1536-3678 (electronic)
DOI:	10.1097/MPH.0000000000002687
Schlagwort:	Male Child Humans Child, Preschool Adolescent Herpesvirus 4, Human Erythrocytes Autoantibodies Cold Temperature Hemoglobinuria, Paroxysmal complications Hemoglobinuria, Paroxysmal diagnosis Epstein-Barr Virus Infections complications Anemia, Hemolytic, Autoimmune diagnosis
Sonstiges:	Nachgewiesen in: MEDLINE Sprachen: English Publication Type: Case Reports; Journal Article Language: English [J Pediatr Hematol Oncol] 2023 Nov 01; Vol. 45 (8), pp. e1014-e1017. <i>Date of Electronic Publication: </i>2023 May 23. MeSH Terms: Hemoglobinuria, Paroxysmal* / complications ; Hemoglobinuria, Paroxysmal* / diagnosis ; Epstein-Barr Virus Infections* / complications ; Anemia, Hemolytic, Autoimmune* / diagnosis ; Male ; Child ; Humans ; Child, Preschool ; Adolescent ; Herpesvirus 4, Human ; Erythrocytes ; Autoantibodies ; Cold Temperature References: Amano S, Kanamori T, Ashiarai M, et al. Severe neutropenia after leukocytosis during an acute attack of mycoplasma infection-associated paroxysmal cold hemoglobinuria. J Pediatr Hematol Oncol. 2017;39:320–321. ; Sokol RJ, Booker DJ, Stamps R. Erythropoiesis: paroxysmal cold haemoglobinuria: a clinico-pathological study of patients with a positive Donath-Landsteiner test. Hematology. 1999;4:137–164. ; Santos Malavé C, Shardy DL, Hartman SK, et al. Paroxysmal cold hemoglobinuria in a child with RSV: an underrecognized cause of hemolytic anemia. Am J Emerg Med. 2016;34:2252.e5–2252.e6. ; Göttsche B, Salama A, Mueller-Eckhardt C. Donath-Landsteiner autoimmune hemolytic anemia in children. A study of 22 cases. Vox Sang. 1990;58:281–286. ; Sankaran J, Rodriguez V, Jacob EK, et al. Autoimmune hemolytic anemia in children: Mayo Clinic experience. J Pediatr Hematol Oncol. 2016;38:e120–e124. ; Shanbhag S, Spivak J. Paroxysmal cold hemoglobinuria. Hematol Oncol Clin N Am. 2015;29:473–478. ; Cooling LLW. Editorial: Kids, colds, and complement: paroxysmal cold hemoglobinuria. Transfusion. 2017;57:1332–1335. ; Vaglio S, Arista MC, Perrone MP, et al. Autoimmune hemolytic anemia in childhood: serologic features in 100 cases. Transfusion. 2017;47:50–54. ; Ghanim MT, Berman B. Immune hemolytic anemia (Paroxysmal Cold Hemoglobinuria) preceding Burkitt lymphoma in a 12-year-old child. J Pediatr Hematol Oncol. 2017;39:e25–e26. ; Wishart MM, Davey MG. Infectious mononucleosis complicated by acute haemolytic anaemia with a positive Donath-Landsteiner reaction. J Clin Pathol. 1973;26:332–334. ; Ogose T, Wakata Y, Kaneko M, et al. A case of recurrent paroxysmal cold hemoglobinuria with the different temperature thresholds of Donath-Landsteiner antibodies. J Pediatr Hematol Oncol. 2007;29:716–719. ; Zeller MP, Arnold DM, Al Habsi K, et al. Paroxysmal cold hemoglobinuria: a difficult diagnosis in adult patients. Transfusion. 2017;57:137–143. ; Lau-Braunhut SA, Stone H, Collins G, et al. Paroxysmal cold hemoglobinuria successfully treated with complement inhibition. Blood Adv. 2019;3:3575–3578. ; Rosse WF, Hillmen P, Schreiber AD. Immune-mediated hemolytic anemia. Am Soc Hematol Rev. 2004:48–62. ; Blackall D, Dolatshahi L. Autoimmune hemolytic anemia in children: Laboratory investigation, disease associations, and treatment strategies. J Pediatr Hematol Oncol. 2022;44:71–78. ; Brugnara C, Barcellini W Post TW. Paroxysmal cold hemoglobinuria UpToDate. UpToDate; 2022. Accessed October 18, 2022. https://www.uptodate.com/contents/paroxysmal-cold-hemoglobinuria. ; Koppel A, Lim S, Osby M, et al. Rituximab as successful therapy in a patient with refractory paroxysmal cold hemoglobinuria. Transfusion. 2007;47:1902–1904. ; Roy-Burman A, Glader BE. Resolution of severe Donath-Landsteiner autoimmune hemolytic anemia temporally associated with institution of plasmapheresis. Crit Care Med. 2002;30:931–934. ; Gregory GP, Opat S, Quach H, et al. Failure of eculizumab to correct paroxysmal cold hemoglobinuria. Ann Hematol. 2011;90:989–990. Substance Nomenclature: 0 (Autoantibodies) Entry Date(s): Date Created: 20230606 Date Completed: 20231030 Latest Revision: 20231101 Update Code: 20240514

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Epstein-Barr Virus-induced Paroxysmal Cold Hemoglobinuria in a 17-Year-old Male.