{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,16]],"date-time":"2026-02-16T21:00:51Z","timestamp":1771275651970,"version":"3.50.1"},"reference-count":22,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2014,9,26]],"date-time":"2014-09-26T00:00:00Z","timestamp":1411689600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Transfusion"],"published-print":{"date-parts":[[2015,3]]},"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Hyperhemolysis is a serious transfusion reaction, most often described in patients with hemoglobinopathies. Hyperhemolysis is characterized by the destruction of host red blood cells (<jats:styled-content style=\"fixed-case\">RBCs<\/jats:styled-content>), in addition to donor<jats:styled-content style=\"fixed-case\">RBCs<\/jats:styled-content>, via an unknown mechanism.<\/jats:p><\/jats:sec><jats:sec><jats:title>Study Design and Methods<\/jats:title><jats:p>We present the case of a 58\u2010year\u2010old woman with treated human immunodeficiency virus and a normal hemoglobin (<jats:styled-content style=\"fixed-case\">Hb<\/jats:styled-content>) electrophoresis who developed hyperhemolysis in the setting of a delayed hemolytic transfusion reaction (<jats:styled-content style=\"fixed-case\">DHTR<\/jats:styled-content>).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The patient was<jats:styled-content style=\"fixed-case\">ABO<\/jats:styled-content>group<jats:styled-content style=\"fixed-case\">B<\/jats:styled-content>and had a previously identified anti\u2010<jats:styled-content style=\"fixed-case\">F<\/jats:styled-content>y<jats:sup>b<\/jats:sup>alloantibody. After transfusion of<jats:styled-content style=\"fixed-case\">F<\/jats:styled-content>y<jats:sup>b<\/jats:sup>\u2013<jats:styled-content style=\"fixed-case\">RBCs<\/jats:styled-content>, she developed a<jats:styled-content style=\"fixed-case\">DHTR<\/jats:styled-content>and was found to have anti\u2010<jats:styled-content style=\"fixed-case\">E<\/jats:styled-content>, anti\u2010<jats:styled-content style=\"fixed-case\">C<\/jats:styled-content><jats:sup>w<\/jats:sup>, anti\u2010s, and an additional antibody to an unrecognized high\u2010frequency<jats:styled-content style=\"fixed-case\">RBC<\/jats:styled-content>alloantigen. Subsequent transfusion of<jats:styled-content style=\"fixed-case\">ABO<\/jats:styled-content>\u2010compatible<jats:styled-content style=\"fixed-case\">RBCs<\/jats:styled-content>that were negative for<jats:styled-content style=\"fixed-case\">F<\/jats:styled-content>y<jats:sup>b<\/jats:sup>,<jats:styled-content style=\"fixed-case\">E<\/jats:styled-content>,<jats:styled-content style=\"fixed-case\">C<\/jats:styled-content><jats:sup>w<\/jats:sup>, and s antigens resulted in immediate intravascular hemolysis. In the absence of bleeding, her hematocrit (<jats:styled-content style=\"fixed-case\">Hct<\/jats:styled-content>) decreased to 10.2%. An extensive serologic evaluation failed to identify the specificity of the high\u2010frequency antibody. Severe hemolytic reactions also occurred despite pretransfusion conditioning with eculizumab. The<jats:styled-content style=\"fixed-case\">Hct<\/jats:styled-content>and clinical symptoms slowly improved after the cessation of transfusions and treatment with erythropoietin and steroids. This case demonstrates several noteworthy features including hyperhemolysis in a patient without a<jats:styled-content style=\"fixed-case\">Hb<\/jats:styled-content>disorder, the development of an antibody to an unknown<jats:styled-content style=\"fixed-case\">RBC<\/jats:styled-content>antigen, and the failure of eculizumab to prevent intravascular hemolysis after transfusion.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Hyperhemolysis is not restricted to patients with hemoglobinopathies. Whether eculizumab offers any benefit in the hyperhemolysis syndrome or in the prevention of intravascular hemolysis due to<jats:styled-content style=\"fixed-case\">RBC<\/jats:styled-content>alloantibodies remains uncertain.<\/jats:p><\/jats:sec>","DOI":"10.1111\/trf.12876","type":"journal-article","created":{"date-parts":[[2014,9,26]],"date-time":"2014-09-26T08:20:14Z","timestamp":1411719614000},"page":"623-628","source":"Crossref","is-referenced-by-count":31,"title":["Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab"],"prefix":"10.1111","volume":"55","author":[{"given":"Shruti","family":"Gupta","sequence":"first","affiliation":[{"name":"Department of Medicine Massachusetts General Hospital Boston Massachusetts"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andrew","family":"Fenves","sequence":"additional","affiliation":[{"name":"Department of Medicine Massachusetts General Hospital Boston Massachusetts"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sandra Taddie","family":"Nance","sequence":"additional","affiliation":[{"name":"American Red Cross Biomedical Services Philadelphia Pennsylvania"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"David B.","family":"Sykes","sequence":"additional","affiliation":[{"name":"Department of Medicine Massachusetts General Hospital Boston Massachusetts"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Walter \u201cSunny\u201d","family":"Dzik","sequence":"additional","affiliation":[{"name":"Department of Medicine Massachusetts General Hospital Boston Massachusetts"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2014,9,26]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1537-2995.2005.00608.x"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1097\/MPH.0b013e31828e529f"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.4103\/0973-6247.45259"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1080\/1024533042000205478"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/tme.12104"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1537-2995.1997.37497265334.x"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1537-2995.2009.02399.x"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.111.6.e661"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1537-2995.1984.24384225018.x"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1537-2995.1997.37497265337.x"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-3148.2011.01131.x"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1080\/10245330400001926"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1537-2995.1997.37497265338.x"},{"key":"e_1_2_7_15_1","first-page":"159","volume-title":"Immune hemolytic anemias","author":"Petz LD","year":"2004"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1537-2995.2008.01693.x"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI110200"},{"key":"e_1_2_7_18_1","doi-asserted-by":"crossref","first-page":"802","DOI":"10.1182\/blood-2006-04-014878","article-title":"Red\u2010cell ICAM\u20104 is a ligand for the monocyte\/macrophage integrin CD11c\/CD18: characterization of the binding sites on ICAM\u20104","volume":"109","author":"Ihanus E","journal-title":"Blood"},{"key":"e_1_2_7_19_1","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1586\/ehm.09.2","article-title":"Hyperhemolysis syndrome in sickle cell disease","volume":"2","author":"Win N","journal-title":"Expert Rev Hematol"},{"key":"e_1_2_7_20_1","doi-asserted-by":"crossref","first-page":"384","DOI":"10.1111\/trf.12258","article-title":"Successful treatment of recurrent hyperhemolysis syndrome with immunosuppression and plasma\u2010to\u2010red blood cell exchange transfusion","volume":"54","author":"Uhlmann EJ","journal-title":"Transfusion"},{"key":"e_1_2_7_21_1","doi-asserted-by":"crossref","first-page":"355","DOI":"10.1111\/j.1423-0410.1995.tb00373.x","article-title":"Post\u2010transfusion hyperhaemolysis in a patient with sickle cell disease: use of steroids and intravenous immunoglobulin to prevent further red cell destruction","volume":"69","author":"Cullis JO","journal-title":"Vox Sang"},{"key":"e_1_2_7_22_1","doi-asserted-by":"crossref","first-page":"1233","DOI":"10.1056\/NEJMoa061648","article-title":"The complement inhibitor eculizumab in paroxysmal nocturnal hemoglobinuria","volume":"355","author":"Hillmen P","journal-title":"N Engl J Med"},{"key":"e_1_2_7_23_1","unstructured":"Alexion Pharmaceuticals Inc.Soliris\u00ae (eculizumab) [package insert]. 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