Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab
Identifieur interne : 001D39 ( Istex/Corpus ); précédent : 001D38; suivant : 001D40Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab
Auteurs : Shruti Gupta ; Andrew Fenves ; Sandra Taddie Nance ; David B. Sykes ; Walter Unny DzikSource :
- Transfusion [ 0041-1132 ] ; 2015-03.
Abstract
Background: Hyperhemolysis is a serious transfusion reaction, most often described in patients with hemoglobinopathies. Hyperhemolysis is characterized by the destruction of host red blood cells (RBCs), in addition to donor RBCs, via an unknown mechanism. Study Design and Methods: We present the case of a 58‐year‐old woman with treated human immunodeficiency virus and a normal hemoglobin (Hb) electrophoresis who developed hyperhemolysis in the setting of a delayed hemolytic transfusion reaction (DHTR). Results: The patient was ABO group B and had a previously identified anti‐Fyb alloantibody. After transfusion of Fyb– RBCs, she developed a DHTR and was found to have anti‐E, anti‐Cw, anti‐s, and an additional antibody to an unrecognized high‐frequency RBC alloantigen. Subsequent transfusion of ABO‐compatible RBCs that were negative for Fyb, E, Cw, and s antigens resulted in immediate intravascular hemolysis. In the absence of bleeding, her hematocrit (Hct) decreased to 10.2%. An extensive serologic evaluation failed to identify the specificity of the high‐frequency antibody. Severe hemolytic reactions also occurred despite pretransfusion conditioning with eculizumab. The Hct 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 Hb disorder, the development of an antibody to an unknown RBC antigen, and the failure of eculizumab to prevent intravascular hemolysis after transfusion. Conclusion: 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 RBC alloantibodies remains uncertain.
Url:
DOI: 10.1111/trf.12876
Links to Exploration step
ISTEX:9126598165B9466030DCB6AD677E6BA3797C48EFLe document en format XML
<record><TEI wicri:istexFullTextTei="biblStruct"><teiHeader><fileDesc><titleStmt><title xml:lang="en">Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab</title><author><name sortKey="Gupta, Shruti" sort="Gupta, Shruti" uniqKey="Gupta S" first="Shruti" last="Gupta">Shruti Gupta</name><affiliation><mods:affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</mods:affiliation></affiliation></author><author><name sortKey="Fenves, Andrew" sort="Fenves, Andrew" uniqKey="Fenves A" first="Andrew" last="Fenves">Andrew Fenves</name><affiliation><mods:affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</mods:affiliation></affiliation></author><author><name sortKey="Nance, Sandra Taddie" sort="Nance, Sandra Taddie" uniqKey="Nance S" first="Sandra Taddie" last="Nance">Sandra Taddie Nance</name><affiliation><mods:affiliation>American Red Cross Biomedical Services, Pennsylvania, Philadelphia</mods:affiliation></affiliation></author><author><name sortKey="Sykes, David B" sort="Sykes, David B" uniqKey="Sykes D" first="David B." last="Sykes">David B. Sykes</name><affiliation><mods:affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</mods:affiliation></affiliation></author><author><name sortKey="Dzik, Walter Unny" sort="Dzik, Walter Unny" uniqKey="Dzik W" first="Walter Unny" last="Dzik">Walter Unny Dzik</name><affiliation><mods:affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</mods:affiliation></affiliation><affiliation><mods:affiliation>E-mail: sdzik@partners.org</mods:affiliation></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">ISTEX</idno><idno type="RBID">ISTEX:9126598165B9466030DCB6AD677E6BA3797C48EF</idno><date when="2015" year="2015">2015</date><idno type="doi">10.1111/trf.12876</idno><idno type="url">https://api.istex.fr/ark:/67375/WNG-13KSVFTT-T/fulltext.pdf</idno><idno type="wicri:Area/Istex/Corpus">001D39</idno><idno type="wicri:explorRef" wicri:stream="Istex" wicri:step="Corpus" wicri:corpus="ISTEX">001D39</idno></publicationStmt><sourceDesc><biblStruct><analytic><title level="a" type="main">Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab</title><author><name sortKey="Gupta, Shruti" sort="Gupta, Shruti" uniqKey="Gupta S" first="Shruti" last="Gupta">Shruti Gupta</name><affiliation><mods:affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</mods:affiliation></affiliation></author><author><name sortKey="Fenves, Andrew" sort="Fenves, Andrew" uniqKey="Fenves A" first="Andrew" last="Fenves">Andrew Fenves</name><affiliation><mods:affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</mods:affiliation></affiliation></author><author><name sortKey="Nance, Sandra Taddie" sort="Nance, Sandra Taddie" uniqKey="Nance S" first="Sandra Taddie" last="Nance">Sandra Taddie Nance</name><affiliation><mods:affiliation>American Red Cross Biomedical Services, Pennsylvania, Philadelphia</mods:affiliation></affiliation></author><author><name sortKey="Sykes, David B" sort="Sykes, David B" uniqKey="Sykes D" first="David B." last="Sykes">David B. Sykes</name><affiliation><mods:affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</mods:affiliation></affiliation></author><author><name sortKey="Dzik, Walter Unny" sort="Dzik, Walter Unny" uniqKey="Dzik W" first="Walter Unny" last="Dzik">Walter Unny Dzik</name><affiliation><mods:affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</mods:affiliation></affiliation><affiliation><mods:affiliation>E-mail: sdzik@partners.org</mods:affiliation></affiliation></author></analytic><monogr></monogr><series><title level="j" type="main">Transfusion</title><title level="j" type="alt">TRANSFUSION</title><idno type="ISSN">0041-1132</idno><idno type="eISSN">1537-2995</idno><imprint><biblScope unit="vol">55</biblScope><biblScope unit="issue">3</biblScope><biblScope unit="page" from="623">623</biblScope><biblScope unit="page" to="628">628</biblScope><biblScope unit="page-count">6</biblScope><date type="published" when="2015-03">2015-03</date></imprint><idno type="ISSN">0041-1132</idno></series></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0041-1132</idno></seriesStmt></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract">Background: Hyperhemolysis is a serious transfusion reaction, most often described in patients with hemoglobinopathies. Hyperhemolysis is characterized by the destruction of host red blood cells (RBCs), in addition to donor RBCs, via an unknown mechanism. Study Design and Methods: We present the case of a 58‐year‐old woman with treated human immunodeficiency virus and a normal hemoglobin (Hb) electrophoresis who developed hyperhemolysis in the setting of a delayed hemolytic transfusion reaction (DHTR). Results: The patient was ABO group B and had a previously identified anti‐Fyb alloantibody. After transfusion of Fyb– RBCs, she developed a DHTR and was found to have anti‐E, anti‐Cw, anti‐s, and an additional antibody to an unrecognized high‐frequency RBC alloantigen. Subsequent transfusion of ABO‐compatible RBCs that were negative for Fyb, E, Cw, and s antigens resulted in immediate intravascular hemolysis. In the absence of bleeding, her hematocrit (Hct) decreased to 10.2%. An extensive serologic evaluation failed to identify the specificity of the high‐frequency antibody. Severe hemolytic reactions also occurred despite pretransfusion conditioning with eculizumab. The Hct 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 Hb disorder, the development of an antibody to an unknown RBC antigen, and the failure of eculizumab to prevent intravascular hemolysis after transfusion. Conclusion: 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 RBC alloantibodies remains uncertain.</div></front></TEI><istex><corpusName>wiley</corpusName><author><json:item><name>Shruti Gupta</name><affiliations><json:string>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</json:string></affiliations></json:item><json:item><name>Andrew Fenves</name><affiliations><json:string>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</json:string></affiliations></json:item><json:item><name>Sandra Taddie Nance</name><affiliations><json:string>American Red Cross Biomedical Services, Pennsylvania, Philadelphia</json:string></affiliations></json:item><json:item><name>David B. Sykes</name><affiliations><json:string>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</json:string></affiliations></json:item><json:item><name>Walter “Sunny” Dzik</name><affiliations><json:string>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</json:string><json:string>E-mail: sdzik@partners.org</json:string></affiliations></json:item></author><articleId><json:string>TRF12876</json:string></articleId><arkIstex>ark:/67375/WNG-13KSVFTT-T</arkIstex><language><json:string>eng</json:string></language><originalGenre><json:string>caseStudy</json:string></originalGenre><abstract>Background: Hyperhemolysis is a serious transfusion reaction, most often described in patients with hemoglobinopathies. Hyperhemolysis is characterized by the destruction of host red blood cells (RBCs), in addition to donor RBCs, via an unknown mechanism. Study Design and Methods: We present the case of a 58‐year‐old woman with treated human immunodeficiency virus and a normal hemoglobin (Hb) electrophoresis who developed hyperhemolysis in the setting of a delayed hemolytic transfusion reaction (DHTR). Results: The patient was ABO group B and had a previously identified anti‐Fyb alloantibody. After transfusion of Fyb– RBCs, she developed a DHTR and was found to have anti‐E, anti‐Cw, anti‐s, and an additional antibody to an unrecognized high‐frequency RBC alloantigen. Subsequent transfusion of ABO‐compatible RBCs that were negative for Fyb, E, Cw, and s antigens resulted in immediate intravascular hemolysis. In the absence of bleeding, her hematocrit (Hct) decreased to 10.2%. An extensive serologic evaluation failed to identify the specificity of the high‐frequency antibody. Severe hemolytic reactions also occurred despite pretransfusion conditioning with eculizumab. The Hct 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 Hb disorder, the development of an antibody to an unknown RBC antigen, and the failure of eculizumab to prevent intravascular hemolysis after transfusion. Conclusion: 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 RBC alloantibodies remains uncertain.</abstract><qualityIndicators><score>8.608</score><pdfWordCount>3608</pdfWordCount><pdfCharCount>23994</pdfCharCount><pdfVersion>1.5</pdfVersion><pdfPageCount>6</pdfPageCount><pdfPageSize>612 x 801.014 pts</pdfPageSize><pdfWordsPerPage>601</pdfWordsPerPage><pdfText>true</pdfText><refBibsNative>true</refBibsNative><abstractWordCount>251</abstractWordCount><abstractCharCount>1812</abstractCharCount><keywordCount>0</keywordCount></qualityIndicators><title>Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab</title><genre><json:string>case-report</json:string></genre><host><title>Transfusion</title><language><json:string>unknown</json:string></language><doi><json:string>10.1111/(ISSN)1537-2995</json:string></doi><issn><json:string>0041-1132</json:string></issn><eissn><json:string>1537-2995</json:string></eissn><publisherId><json:string>TRF</json:string></publisherId><volume>55</volume><issue>3</issue><pages><first>623</first><last>628</last><total>6</total></pages><genre><json:string>journal</json:string></genre><subject><json:item><value>TRANSFUSION PRACTICE</value></json:item><json:item><value>TRANSFUSION PRACTICE</value></json:item></subject></host><ark><json:string>ark:/67375/WNG-13KSVFTT-T</json:string></ark><categories><wos><json:string>1 - science</json:string><json:string>2 - hematology</json:string></wos><scienceMetrix><json:string>1 - health sciences</json:string><json:string>2 - clinical medicine</json:string><json:string>3 - cardiovascular system & hematology</json:string></scienceMetrix><scopus><json:string>1 - Health Sciences</json:string><json:string>2 - Medicine</json:string><json:string>3 - Hematology</json:string><json:string>1 - Life Sciences</json:string><json:string>2 - Immunology and Microbiology</json:string><json:string>3 - Immunology</json:string><json:string>1 - Health Sciences</json:string><json:string>2 - Medicine</json:string><json:string>3 - Immunology and Allergy</json:string></scopus><inist><json:string>1 - sciences appliquees, technologies et medecines</json:string><json:string>2 - sciences biologiques et medicales</json:string><json:string>3 - sciences medicales</json:string></inist></categories><publicationDate>2015</publicationDate><copyrightDate>2015</copyrightDate><doi><json:string>10.1111/trf.12876</json:string></doi><id>9126598165B9466030DCB6AD677E6BA3797C48EF</id><score>1</score><fulltext><json:item><extension>pdf</extension><original>true</original><mimetype>application/pdf</mimetype><uri>https://api.istex.fr/ark:/67375/WNG-13KSVFTT-T/fulltext.pdf</uri></json:item><json:item><extension>zip</extension><original>false</original><mimetype>application/zip</mimetype><uri>https://api.istex.fr/ark:/67375/WNG-13KSVFTT-T/bundle.zip</uri></json:item><istex:fulltextTEI uri="https://api.istex.fr/ark:/67375/WNG-13KSVFTT-T/fulltext.tei"><teiHeader><fileDesc><titleStmt><title level="a" type="main">Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab</title><title level="a" type="short">Hyperhemolysis</title></titleStmt><publicationStmt><authority>ISTEX</authority><publisher ref="https://scientific-publisher.data.istex.fr/ark:/67375/H02-QW5Q88H5-V">Wiley Publishing Ltd</publisher><availability><licence>© 2014 AABB</licence></availability><date type="published" when="2015-03"></date></publicationStmt><notesStmt><note type="content-type" subtype="case-report" source="caseStudy" scheme="https://content-type.data.istex.fr/ark:/67375/XTP-29919SZJ-6">case-report</note><note type="publication-type" subtype="journal" scheme="https://publication-type.data.istex.fr/ark:/67375/JMC-0GLKJH51-B">journal</note></notesStmt><sourceDesc><biblStruct type="case-report"><analytic><title level="a" type="main">Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab</title><title level="a" type="short">Hyperhemolysis</title><author xml:id="author-0000"><persName><forename type="first">Shruti</forename><surname>Gupta</surname></persName><affiliation><orgName type="division">Department of Medicine</orgName><orgName type="institution">Massachusetts General Hospital</orgName><address><settlement>Boston</settlement><region>Massachusetts</region></address></affiliation></author><author xml:id="author-0001"><persName><forename type="first">Andrew</forename><surname>Fenves</surname></persName><affiliation><orgName type="division">Department of Medicine</orgName><orgName type="institution">Massachusetts General Hospital</orgName><address><settlement>Boston</settlement><region>Massachusetts</region></address></affiliation></author><author xml:id="author-0002"><persName><forename type="first">Sandra Taddie</forename><surname>Nance</surname></persName><affiliation><orgName type="institution">American Red Cross Biomedical Services</orgName><address><settlement>Philadelphia</settlement><region>Pennsylvania</region></address></affiliation></author><author xml:id="author-0003"><persName><forename type="first">David B.</forename><surname>Sykes</surname></persName><affiliation><orgName type="division">Department of Medicine</orgName><orgName type="institution">Massachusetts General Hospital</orgName><address><settlement>Boston</settlement><region>Massachusetts</region></address></affiliation></author><author xml:id="author-0004" role="corresp"><persName><forename type="first">Walter “Sunny”</forename><surname>Dzik</surname></persName><affiliation><orgName type="division">Department of Medicine</orgName><orgName type="institution">Massachusetts General Hospital</orgName><address><settlement>Boston</settlement><region>Massachusetts</region></address></affiliation></author><idno type="istex">9126598165B9466030DCB6AD677E6BA3797C48EF</idno><idno type="ark">ark:/67375/WNG-13KSVFTT-T</idno><idno type="DOI">10.1111/trf.12876</idno><idno type="unit">TRF12876</idno><idno type="toTypesetVersion">file:TRF.TRF12876.pdf</idno></analytic><monogr><title level="j" type="main">Transfusion</title><title level="j" type="alt">TRANSFUSION</title><idno type="pISSN">0041-1132</idno><idno type="eISSN">1537-2995</idno><idno type="book-DOI">10.1111/(ISSN)1537-2995</idno><idno type="book-part-DOI">10.1111/trf.v55.3</idno><idno type="product">TRF</idno><imprint><biblScope unit="vol">55</biblScope><biblScope unit="issue">3</biblScope><biblScope unit="page" from="623">623</biblScope><biblScope unit="page" to="628">628</biblScope><biblScope unit="page-count">6</biblScope><date type="published" when="2015-03"></date></imprint></monogr></biblStruct></sourceDesc></fileDesc><encodingDesc><schemaRef type="ODD" url="https://xml-schema.delivery.istex.fr/tei-istex.odd"></schemaRef><appInfo><application ident="pub2tei" version="1.0.10" when="2019-12-20"><label>pub2TEI-ISTEX</label><desc>A set of style sheets for converting XML documents encoded in various scientific publisher formats into a common TEI format.
<ref target="http://www.tei-c.org/">We use TEI</ref></desc></application></appInfo></encodingDesc><profileDesc><abstract style="main"><head>Background</head><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 (<hi rend="fc">RBCs</hi>), in addition to donor <hi rend="fc">RBCs</hi>, via an unknown mechanism.</p><head>Study Design and Methods</head><p>We present the case of a 58‐year‐old woman with treated human immunodeficiency virus and a normal hemoglobin (<hi rend="fc">Hb</hi>) electrophoresis who developed hyperhemolysis in the setting of a delayed hemolytic transfusion reaction (<hi rend="fc">DHTR</hi>).</p><head>Results</head><p>The patient was <hi rend="fc">ABO</hi> group <hi rend="fc">B</hi> and had a previously identified anti‐<hi rend="fc">F</hi>y<hi rend="superscript">b</hi> alloantibody. After transfusion of <hi rend="fc">F</hi>y<hi rend="superscript">b</hi>– <hi rend="fc">RBCs</hi>, she developed a <hi rend="fc">DHTR</hi> and was found to have anti‐<hi rend="fc">E</hi>, anti‐<hi rend="fc">C</hi><hi rend="superscript">w</hi>, anti‐s, and an additional antibody to an unrecognized high‐frequency <hi rend="fc">RBC</hi> alloantigen. Subsequent transfusion of <hi rend="fc">ABO</hi>‐compatible <hi rend="fc">RBCs</hi> that were negative for <hi rend="fc">F</hi>y<hi rend="superscript">b</hi>, <hi rend="fc">E</hi>, <hi rend="fc">C</hi><hi rend="superscript">w</hi>, and s antigens resulted in immediate intravascular hemolysis. In the absence of bleeding, her hematocrit (<hi rend="fc">Hct</hi>) decreased to 10.2%. An extensive serologic evaluation failed to identify the specificity of the high‐frequency antibody. Severe hemolytic reactions also occurred despite pretransfusion conditioning with eculizumab. The <hi rend="fc">Hct</hi> 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 <hi rend="fc">Hb</hi> disorder, the development of an antibody to an unknown <hi rend="fc">RBC</hi> antigen, and the failure of eculizumab to prevent intravascular hemolysis after transfusion.</p><head>Conclusion</head><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 <hi rend="fc">RBC</hi> alloantibodies remains uncertain.</p></abstract><textClass><keywords rend="articleCategory"><term>TRANSFUSION PRACTICE</term></keywords><keywords rend="tocHeading1"><term>TRANSFUSION PRACTICE</term></keywords></textClass><langUsage><language ident="en"></language></langUsage></profileDesc><revisionDesc><change when="2019-12-20" who="#istex" xml:id="pub2tei">formatting</change></revisionDesc></teiHeader></istex:fulltextTEI><json:item><extension>txt</extension><original>false</original><mimetype>text/plain</mimetype><uri>https://api.istex.fr/ark:/67375/WNG-13KSVFTT-T/fulltext.txt</uri></json:item></fulltext><metadata><istex:metadataXml wicri:clean="Wiley, elements deleted: body"><istex:xmlDeclaration>version="1.0" encoding="UTF-8" standalone="yes"</istex:xmlDeclaration><istex:document><component type="serialArticle" version="2.0" xml:id="trf12876" xml:lang="en"><header><publicationMeta level="product"><doi origin="wiley">10.1111/(ISSN)1537-2995</doi><issn type="print">0041-1132</issn><issn type="electronic">1537-2995</issn><idGroup><id type="product" value="TRF"></id></idGroup><titleGroup><title sort="TRANSFUSION" type="main">Transfusion</title><title type="short">Transfusion</title></titleGroup></publicationMeta><publicationMeta level="part" position="30"><doi>10.1111/trf.v55.3</doi><copyright ownership="thirdParty">© 2015 AABB</copyright><numberingGroup><numbering type="journalVolume" number="55">55</numbering><numbering type="journalIssue">3</numbering></numberingGroup><coverDate startDate="2015-03">March 2015</coverDate></publicationMeta><publicationMeta level="unit" position="240" status="forIssue" type="caseStudy"><doi>10.1111/trf.12876</doi><idGroup><id type="unit" value="TRF12876"></id></idGroup><countGroup><count number="6" type="pageTotal"></count></countGroup><titleGroup><title type="articleCategory">TRANSFUSION PRACTICE</title><title type="tocHeading1">TRANSFUSION PRACTICE</title></titleGroup><copyright ownership="thirdParty">© 2014 AABB</copyright><eventGroup><event agent="bestset" date="2014-09-05" type="xmlCreated"></event><event date="2014-06-29" type="manuscriptReceived"></event><event date="2014-08-11" type="manuscriptRevised"></event><event date="2014-08-11" type="manuscriptAccepted"></event><event type="publishedOnlineEarlyUnpaginated" date="2014-09-26"></event><event type="firstOnline" date="2014-09-26"></event><event type="publishedOnlineFinalForm" date="2015-03-11"></event><event type="xmlConverted" agent="Converter:WML3G_To_WML3G version:4.7.2 mode:FullText" date="2016-01-06"></event></eventGroup><numberingGroup><numbering type="pageFirst">623</numbering><numbering type="pageLast">628</numbering></numberingGroup><correspondenceTo><i>Address reprint requests to</i>: Walter H. Dzik, MD, Blood Transfusion Service, J‐224, Massachusetts General Hospital, Boston, MA 02114; e‐mail: <email>sdzik@partners.org</email>.</correspondenceTo><linkGroup><link type="toTypesetVersion" href="file:TRF.TRF12876.pdf"></link></linkGroup></publicationMeta><contentMeta><titleGroup><title type="main">Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab</title><title type="short">Hyperhemolysis</title><title type="shortAuthors">Gupta et al.</title></titleGroup><creators><creator affiliationRef="#trf12876-aff-0001" creatorRole="author" xml:id="trf12876-cr-0001"><personName><givenNames>Shruti</givenNames><familyName>Gupta</familyName></personName></creator><creator affiliationRef="#trf12876-aff-0001" creatorRole="author" xml:id="trf12876-cr-0002"><personName><givenNames>Andrew</givenNames><familyName>Fenves</familyName></personName></creator><creator affiliationRef="#trf12876-aff-0002" creatorRole="author" xml:id="trf12876-cr-0003"><personName><givenNames>Sandra Taddie</givenNames><familyName>Nance</familyName></personName></creator><creator affiliationRef="#trf12876-aff-0001" creatorRole="author" xml:id="trf12876-cr-0004"><personName><givenNames>David B.</givenNames><familyName>Sykes</familyName></personName></creator><creator affiliationRef="#trf12876-aff-0001" corresponding="yes" creatorRole="author" xml:id="trf12876-cr-0005"><personName><givenNames>Walter “Sunny”</givenNames><familyName>Dzik</familyName></personName></creator></creators><affiliationGroup><affiliation xml:id="trf12876-aff-0001"><orgDiv>Department of Medicine</orgDiv><orgName>Massachusetts General Hospital</orgName><address><city>Boston</city><countryPart>Massachusetts</countryPart></address></affiliation><affiliation xml:id="trf12876-aff-0002"><orgName>American Red Cross Biomedical Services</orgName><address><city>Philadelphia</city><countryPart>Pennsylvania</countryPart></address></affiliation></affiliationGroup><abstractGroup><abstract type="main"><section xml:id="trf12876-sec-0001"><title type="main">Background</title><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 (<fc>RBCs</fc>), in addition to donor <fc>RBCs</fc>, via an unknown mechanism.</p></section><section xml:id="trf12876-sec-0002"><title type="main">Study Design and Methods</title><p>We present the case of a 58‐year‐old woman with treated human immunodeficiency virus and a normal hemoglobin (<fc>Hb</fc>) electrophoresis who developed hyperhemolysis in the setting of a delayed hemolytic transfusion reaction (<fc>DHTR</fc>).</p></section><section xml:id="trf12876-sec-0003"><title type="main">Results</title><p>The patient was <fc>ABO</fc> group <fc>B</fc> and had a previously identified anti‐<fc>F</fc>y<sup>b</sup> alloantibody. After transfusion of <fc>F</fc>y<sup>b</sup>– <fc>RBCs</fc>, she developed a <fc>DHTR</fc> and was found to have anti‐<fc>E</fc>, anti‐<fc>C</fc><sup>w</sup>, anti‐s, and an additional antibody to an unrecognized high‐frequency <fc>RBC</fc> alloantigen. Subsequent transfusion of <fc>ABO</fc>‐compatible <fc>RBCs</fc> that were negative for <fc>F</fc>y<sup>b</sup>, <fc>E</fc>, <fc>C</fc><sup>w</sup>, and s antigens resulted in immediate intravascular hemolysis. In the absence of bleeding, her hematocrit (<fc>Hct</fc>) decreased to 10.2%. An extensive serologic evaluation failed to identify the specificity of the high‐frequency antibody. Severe hemolytic reactions also occurred despite pretransfusion conditioning with eculizumab. The <fc>Hct</fc> 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 <fc>Hb</fc> disorder, the development of an antibody to an unknown <fc>RBC</fc> antigen, and the failure of eculizumab to prevent intravascular hemolysis after transfusion.</p></section><section xml:id="trf12876-sec-0004"><title type="main">Conclusion</title><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 <fc>RBC</fc> alloantibodies remains uncertain.</p></section></abstract></abstractGroup></contentMeta></header></component></istex:document></istex:metadataXml><mods version="3.6"><titleInfo lang="en"><title>Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab</title></titleInfo><titleInfo type="abbreviated" lang="en"><title>Hyperhemolysis</title></titleInfo><titleInfo type="alternative" contentType="CDATA" lang="en"><title>Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab</title></titleInfo><name type="personal"><namePart type="given">Shruti</namePart><namePart type="family">Gupta</namePart><affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Andrew</namePart><namePart type="family">Fenves</namePart><affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Sandra Taddie</namePart><namePart type="family">Nance</namePart><affiliation>American Red Cross Biomedical Services, Pennsylvania, Philadelphia</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">David B.</namePart><namePart type="family">Sykes</namePart><affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Walter “Sunny”</namePart><namePart type="family">Dzik</namePart><affiliation>Department of Medicine, Massachusetts General Hospital, Massachusetts, Boston</affiliation><affiliation>E-mail: sdzik@partners.org</affiliation><role><roleTerm type="text">author</roleTerm></role></name><typeOfResource>text</typeOfResource><genre type="case-report" displayLabel="caseStudy" authority="ISTEX" authorityURI="https://content-type.data.istex.fr" valueURI="https://content-type.data.istex.fr/ark:/67375/XTP-29919SZJ-6">case-report</genre><originInfo><publisher>Blackwell Publishing Ltd</publisher><dateIssued encoding="w3cdtf">2015-03</dateIssued><dateCreated encoding="w3cdtf">2014-09-05</dateCreated><dateCaptured encoding="w3cdtf">2014-06-29</dateCaptured><dateValid encoding="w3cdtf">2014-08-11</dateValid><copyrightDate encoding="w3cdtf">2015</copyrightDate></originInfo><language><languageTerm type="code" authority="rfc3066">en</languageTerm><languageTerm type="code" authority="iso639-2b">eng</languageTerm></language><abstract>Background: Hyperhemolysis is a serious transfusion reaction, most often described in patients with hemoglobinopathies. Hyperhemolysis is characterized by the destruction of host red blood cells (RBCs), in addition to donor RBCs, via an unknown mechanism. Study Design and Methods: We present the case of a 58‐year‐old woman with treated human immunodeficiency virus and a normal hemoglobin (Hb) electrophoresis who developed hyperhemolysis in the setting of a delayed hemolytic transfusion reaction (DHTR). Results: The patient was ABO group B and had a previously identified anti‐Fyb alloantibody. After transfusion of Fyb– RBCs, she developed a DHTR and was found to have anti‐E, anti‐Cw, anti‐s, and an additional antibody to an unrecognized high‐frequency RBC alloantigen. Subsequent transfusion of ABO‐compatible RBCs that were negative for Fyb, E, Cw, and s antigens resulted in immediate intravascular hemolysis. In the absence of bleeding, her hematocrit (Hct) decreased to 10.2%. An extensive serologic evaluation failed to identify the specificity of the high‐frequency antibody. Severe hemolytic reactions also occurred despite pretransfusion conditioning with eculizumab. The Hct 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 Hb disorder, the development of an antibody to an unknown RBC antigen, and the failure of eculizumab to prevent intravascular hemolysis after transfusion. Conclusion: 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 RBC alloantibodies remains uncertain.</abstract><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><titleInfo type="abbreviated"><title>Transfusion</title></titleInfo><genre type="journal" authority="ISTEX" authorityURI="https://publication-type.data.istex.fr" valueURI="https://publication-type.data.istex.fr/ark:/67375/JMC-0GLKJH51-B">journal</genre><subject><genre>article-category</genre><topic>TRANSFUSION PRACTICE</topic><topic>TRANSFUSION PRACTICE</topic></subject><identifier type="ISSN">0041-1132</identifier><identifier type="eISSN">1537-2995</identifier><identifier type="DOI">10.1111/(ISSN)1537-2995</identifier><identifier type="PublisherID">TRF</identifier><part><date>2015</date><detail type="volume"><caption>vol.</caption><number>55</number></detail><detail type="issue"><caption>no.</caption><number>3</number></detail><extent unit="pages"><start>623</start><end>628</end><total>6</total></extent></part></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0001"><titleInfo><title>Hyperhemolysis syndrome in anemia of chronic disease</title></titleInfo><name type="personal"><namePart type="given">D</namePart><namePart type="family">Darabi</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">W</namePart><namePart type="family">Dzik</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Darabi D, Dzik W. Hyperhemolysis syndrome in anemia of chronic disease. Transfusion 2005;45:1930‐1933.</note><part><date>2005</date><detail type="volume"><caption>vol.</caption><number>45</number></detail><extent unit="pages"><start>1930</start><end>1933</end></extent></part><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><part><date>2005</date><detail type="volume"><caption>vol.</caption><number>45</number></detail><extent unit="pages"><start>1930</start><end>1933</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0002"><titleInfo><title>Hyperhemolysis in sickle cell disease</title></titleInfo><name type="personal"><namePart type="given">E</namePart><namePart type="family">Aragona</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">MJ</namePart><namePart type="family">Kelly</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Aragona E, Kelly MJ. Hyperhemolysis in sickle cell disease. J Pediatr Hematol Oncol 2014;36:e54‐56.</note><part><date>2014</date><detail type="volume"><caption>vol.</caption><number>36</number></detail><extent unit="pages"><start>e54</start><end>56</end></extent></part><relatedItem type="host"><titleInfo><title>J Pediatr Hematol Oncol</title></titleInfo><part><date>2014</date><detail type="volume"><caption>vol.</caption><number>36</number></detail><extent unit="pages"><start>e54</start><end>56</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0003"><titleInfo><title>Hyperhemolysis in a patient with beta‐thalassemia major</title></titleInfo><name type="personal"><namePart type="given">LR</namePart><namePart type="family">Morawakage</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">BJ</namePart><namePart type="family">Perera</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">PD</namePart><namePart type="family">Dias</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Morawakage LR, Perera BJ, Dias PD, et al. Hyperhemolysis in a patient with beta‐thalassemia major. Asian J Transfus Sci 2009;3:26‐27.</note><part><date>2009</date><detail type="volume"><caption>vol.</caption><number>3</number></detail><extent unit="pages"><start>26</start><end>27</end></extent></part><relatedItem type="host"><titleInfo><title>Asian J Transfus Sci</title></titleInfo><part><date>2009</date><detail type="volume"><caption>vol.</caption><number>3</number></detail><extent unit="pages"><start>26</start><end>27</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0004"><titleInfo><title>Hyperhaemolysis syndrome in a patient with myelofibrosis</title></titleInfo><name type="personal"><namePart type="given">JG</namePart><namePart type="family">Treleaven</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">N</namePart><namePart type="family">Win</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Treleaven JG, Win N. Hyperhaemolysis syndrome in a patient with myelofibrosis. Hematology 2004;9:147‐149.</note><part><date>2004</date><detail type="volume"><caption>vol.</caption><number>9</number></detail><extent unit="pages"><start>147</start><end>149</end></extent></part><relatedItem type="host"><titleInfo><title>Hematology</title></titleInfo><part><date>2004</date><detail type="volume"><caption>vol.</caption><number>9</number></detail><extent unit="pages"><start>147</start><end>149</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0005"><titleInfo><title>Hyperhaemolysis in a patient with chronic lymphocytic leukaemia</title></titleInfo><name type="personal"><namePart type="given">M</namePart><namePart type="family">Rogers</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">G</namePart><namePart type="family">Smith</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Rogers M, Smith G. Hyperhaemolysis in a patient with chronic lymphocytic leukaemia. Transfus Med 2014;24:123‐124.</note><part><date>2014</date><detail type="volume"><caption>vol.</caption><number>24</number></detail><extent unit="pages"><start>123</start><end>124</end></extent></part><relatedItem type="host"><titleInfo><title>Transfus Med</title></titleInfo><part><date>2014</date><detail type="volume"><caption>vol.</caption><number>24</number></detail><extent unit="pages"><start>123</start><end>124</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0006"><titleInfo><title>Severe reactions associated with transfusion of patients with sickle cell disease</title></titleInfo><name type="personal"><namePart type="given">G</namePart><namePart type="family">Garratty</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Garratty G. Severe reactions associated with transfusion of patients with sickle cell disease. Transfusion 1997;37:351‐361.</note><part><date>1997</date><detail type="volume"><caption>vol.</caption><number>37</number></detail><extent unit="pages"><start>351</start><end>361</end></extent></part><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><part><date>1997</date><detail type="volume"><caption>vol.</caption><number>37</number></detail><extent unit="pages"><start>351</start><end>361</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0007"><titleInfo><title>Delayed hemolytic transfusion reaction with hyperhemolysis after first red blood cell transfusion in child with beta‐thalassemia: challenges in treatment</title></titleInfo><name type="personal"><namePart type="given">SE</namePart><namePart type="family">Hannema</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">A</namePart><namePart type="family">Brand</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">A</namePart><namePart type="family">van Meurs</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Hannema SE, Brand A, van Meurs A, et al. Delayed hemolytic transfusion reaction with hyperhemolysis after first red blood cell transfusion in child with beta‐thalassemia: challenges in treatment. Transfusion 2010;50:429‐432.</note><part><date>2010</date><detail type="volume"><caption>vol.</caption><number>50</number></detail><extent unit="pages"><start>429</start><end>432</end></extent></part><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><part><date>2010</date><detail type="volume"><caption>vol.</caption><number>50</number></detail><extent unit="pages"><start>429</start><end>432</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0008"><titleInfo><title>Delayed hemolytic transfusion reaction/hyperhemolysis syndrome in children with sickle cell disease</title></titleInfo><name type="personal"><namePart type="given">JA</namePart><namePart type="family">Talano</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">CA</namePart><namePart type="family">Hillery</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">JL</namePart><namePart type="family">Gottschall</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Talano JA, Hillery CA, Gottschall JL, et al. Delayed hemolytic transfusion reaction/hyperhemolysis syndrome in children with sickle cell disease. Pediatrics 2003;111:661‐663.</note><part><date>2003</date><detail type="volume"><caption>vol.</caption><number>111</number></detail><extent unit="pages"><start>661</start><end>663</end></extent></part><relatedItem type="host"><titleInfo><title>Pediatrics</title></titleInfo><part><date>2003</date><detail type="volume"><caption>vol.</caption><number>111</number></detail><extent unit="pages"><start>661</start><end>663</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0009"><titleInfo><title>Delayed hemolytic transfusion reactions. Evidence for complement activation involving allogeneic and autologous red cells</title></titleInfo><name type="personal"><namePart type="given">A</namePart><namePart type="family">Salama</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">C</namePart><namePart type="family">Mueller‐Eckhardt</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Salama A, Mueller‐Eckhardt C. Delayed hemolytic transfusion reactions. Evidence for complement activation involving allogeneic and autologous red cells. Transfusion 1984;24:188‐193.</note><part><date>1984</date><detail type="volume"><caption>vol.</caption><number>24</number></detail><extent unit="pages"><start>188</start><end>193</end></extent></part><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><part><date>1984</date><detail type="volume"><caption>vol.</caption><number>24</number></detail><extent unit="pages"><start>188</start><end>193</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0010"><titleInfo><title>Delayed hemolytic transfusion reactions in sickle cell disease: simultaneous destruction of recipients' red cells</title></titleInfo><name type="personal"><namePart type="given">KE</namePart><namePart type="family">King</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">RS</namePart><namePart type="family">Shirey</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">MW</namePart><namePart type="family">Lankiewicz</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">King KE, Shirey RS, Lankiewicz MW, et al. Delayed hemolytic transfusion reactions in sickle cell disease: simultaneous destruction of recipients' red cells. Transfusion 1997;37:376‐381.</note><part><date>1997</date><detail type="volume"><caption>vol.</caption><number>37</number></detail><extent unit="pages"><start>376</start><end>381</end></extent></part><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><part><date>1997</date><detail type="volume"><caption>vol.</caption><number>37</number></detail><extent unit="pages"><start>376</start><end>381</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0011"><titleInfo><title>Measurement of macrophage marker in hyperhaemolytic transfusion reaction: a case report</title></titleInfo><name type="personal"><namePart type="given">N</namePart><namePart type="family">Win</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">E</namePart><namePart type="family">Lee</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">M</namePart><namePart type="family">Needs</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Win N, Lee E, Needs M, et al. Measurement of macrophage marker in hyperhaemolytic transfusion reaction: a case report. Transfus Med 2012;22:137‐141.</note><part><date>2012</date><detail type="volume"><caption>vol.</caption><number>22</number></detail><extent unit="pages"><start>137</start><end>141</end></extent></part><relatedItem type="host"><titleInfo><title>Transfus Med</title></titleInfo><part><date>2012</date><detail type="volume"><caption>vol.</caption><number>22</number></detail><extent unit="pages"><start>137</start><end>141</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0012"><titleInfo><title>Use of intravenous immunoglobulin and intravenous methylprednisolone in hyperhaemolysis syndrome in sickle cell disease</title></titleInfo><name type="personal"><namePart type="given">N</namePart><namePart type="family">Win</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">T</namePart><namePart type="family">Yeghen</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">M</namePart><namePart type="family">Needs</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Win N, Yeghen T, Needs M, et al. Use of intravenous immunoglobulin and intravenous methylprednisolone in hyperhaemolysis syndrome in sickle cell disease. Hematology 2004;9:433‐436.</note><part><date>2004</date><detail type="volume"><caption>vol.</caption><number>9</number></detail><extent unit="pages"><start>433</start><end>436</end></extent></part><relatedItem type="host"><titleInfo><title>Hematology</title></titleInfo><part><date>2004</date><detail type="volume"><caption>vol.</caption><number>9</number></detail><extent unit="pages"><start>433</start><end>436</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0013"><titleInfo><title>The sickle cell hemolytic transfusion reaction syndrome</title></titleInfo><name type="personal"><namePart type="given">LD</namePart><namePart type="family">Petz</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Petz LD. The sickle cell hemolytic transfusion reaction syndrome. Transfusion 1997;37:382‐392.</note><part><date>1997</date><detail type="volume"><caption>vol.</caption><number>37</number></detail><extent unit="pages"><start>382</start><end>392</end></extent></part><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><part><date>1997</date><detail type="volume"><caption>vol.</caption><number>37</number></detail><extent unit="pages"><start>382</start><end>392</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0014"><titleInfo><title>Petz LD, Garratty G. Immune hemolytic anemias. 2nd ed. Philadelphia (PA): Churchill Livingstone; 2004. p. 159‐160.</title></titleInfo><note type="citation/reference">Petz LD, Garratty G. Immune hemolytic anemias. 2nd ed. Philadelphia (PA): Churchill Livingstone; 2004. p. 159‐160.</note><name type="personal"><namePart type="given">LD</namePart><namePart type="family">Petz</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">G</namePart><namePart type="family">Garratty</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>book</genre><originInfo><publisher>Churchill Livingstone</publisher><place><placeTerm type="text">Philadelphia (PA)</placeTerm></place></originInfo><part><date>2004</date><extent unit="pages"><start>159</start><end>160</end></extent></part></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0015"><titleInfo><title>Hyperhemolysis syndrome in sickle cell disease: a case report (recurrent episode) and literature review</title></titleInfo><name type="personal"><namePart type="given">N</namePart><namePart type="family">Win</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">H</namePart><namePart type="family">New</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">E</namePart><namePart type="family">Lee</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Win N, New H, Lee E, et al. Hyperhemolysis syndrome in sickle cell disease: a case report (recurrent episode) and literature review. Transfusion 2008;48:1231‐1238.</note><part><date>2008</date><detail type="volume"><caption>vol.</caption><number>48</number></detail><extent unit="pages"><start>1231</start><end>1238</end></extent></part><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><part><date>2008</date><detail type="volume"><caption>vol.</caption><number>48</number></detail><extent unit="pages"><start>1231</start><end>1238</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0016"><titleInfo><title>Abnormalities in membrane phospholipid organization in sickled erythrocytes</title></titleInfo><name type="personal"><namePart type="given">B</namePart><namePart type="family">Lubin</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">D</namePart><namePart type="family">Chiu</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">J</namePart><namePart type="family">Bastacky</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Lubin B, Chiu D, Bastacky J, et al. Abnormalities in membrane phospholipid organization in sickled erythrocytes. J Clin Invest 1981;67:1643‐1649.</note><part><date>1981</date><detail type="volume"><caption>vol.</caption><number>67</number></detail><extent unit="pages"><start>1643</start><end>1649</end></extent></part><relatedItem type="host"><titleInfo><title>J Clin Invest</title></titleInfo><part><date>1981</date><detail type="volume"><caption>vol.</caption><number>67</number></detail><extent unit="pages"><start>1643</start><end>1649</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0017"><titleInfo><title>Red‐cell ICAM‐4 is a ligand for the monocyte/macrophage integrin CD11c/CD18: characterization of the binding sites on ICAM‐4</title></titleInfo><name type="personal"><namePart type="given">E</namePart><namePart type="family">Ihanus</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">LM</namePart><namePart type="family">Uotila</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">A</namePart><namePart type="family">Toivanen</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Ihanus E, Uotila LM, Toivanen A, et al. Red‐cell ICAM‐4 is a ligand for the monocyte/macrophage integrin CD11c/CD18: characterization of the binding sites on ICAM‐4. Blood 2007;109:802‐810.</note><part><detail type="volume"><caption>vol.</caption><number>109</number></detail><extent unit="pages"><start>802</start><end>810</end></extent></part><relatedItem type="host"><titleInfo><title>Blood</title></titleInfo><part><detail type="volume"><caption>vol.</caption><number>109</number></detail><extent unit="pages"><start>802</start><end>810</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0018"><titleInfo><title>Hyperhemolysis syndrome in sickle cell disease</title></titleInfo><name type="personal"><namePart type="given">N</namePart><namePart type="family">Win</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Win N. Hyperhemolysis syndrome in sickle cell disease. Expert Rev Hematol 2009;2:111‐115.</note><part><detail type="volume"><caption>vol.</caption><number>2</number></detail><extent unit="pages"><start>111</start><end>115</end></extent></part><relatedItem type="host"><titleInfo><title>Expert Rev Hematol</title></titleInfo><part><detail type="volume"><caption>vol.</caption><number>2</number></detail><extent unit="pages"><start>111</start><end>115</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0019"><titleInfo><title>Successful treatment of recurrent hyperhemolysis syndrome with immunosuppression and plasma‐to‐red blood cell exchange transfusion</title></titleInfo><name type="personal"><namePart type="given">EJ</namePart><namePart type="family">Uhlmann</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">S</namePart><namePart type="family">Shenoy</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">LT</namePart><namePart type="family">Goodnough</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Uhlmann EJ, Shenoy S, Goodnough LT. Successful treatment of recurrent hyperhemolysis syndrome with immunosuppression and plasma‐to‐red blood cell exchange transfusion. Transfusion 2014;54:384‐388.</note><part><detail type="volume"><caption>vol.</caption><number>54</number></detail><extent unit="pages"><start>384</start><end>388</end></extent></part><relatedItem type="host"><titleInfo><title>Transfusion</title></titleInfo><part><detail type="volume"><caption>vol.</caption><number>54</number></detail><extent unit="pages"><start>384</start><end>388</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0020"><titleInfo><title>Post‐transfusion hyperhaemolysis in a patient with sickle cell disease: use of steroids and intravenous immunoglobulin to prevent further red cell destruction</title></titleInfo><name type="personal"><namePart type="given">JO</namePart><namePart type="family">Cullis</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">N</namePart><namePart type="family">Win</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">JM</namePart><namePart type="family">Dudley</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Cullis JO, Win N, Dudley JM, et al. Post‐transfusion hyperhaemolysis in a patient with sickle cell disease: use of steroids and intravenous immunoglobulin to prevent further red cell destruction. Vox Sang 1995;69:355‐357.</note><part><detail type="volume"><caption>vol.</caption><number>69</number></detail><extent unit="pages"><start>355</start><end>357</end></extent></part><relatedItem type="host"><titleInfo><title>Vox Sang</title></titleInfo><part><detail type="volume"><caption>vol.</caption><number>69</number></detail><extent unit="pages"><start>355</start><end>357</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0021"><titleInfo><title>The complement inhibitor eculizumab in paroxysmal nocturnal hemoglobinuria</title></titleInfo><name type="personal"><namePart type="given">P</namePart><namePart type="family">Hillmen</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">NS</namePart><namePart type="family">Young</namePart><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">J</namePart><namePart type="family">Schubert</namePart><role><roleTerm type="text">author</roleTerm></role></name><genre>journal-article</genre><note type="citation/reference">Hillmen P, Young NS, Schubert J, et al. The complement inhibitor eculizumab in paroxysmal nocturnal hemoglobinuria. N Engl J Med 2006;355:1233‐1243.</note><part><detail type="volume"><caption>vol.</caption><number>355</number></detail><extent unit="pages"><start>1233</start><end>1243</end></extent></part><relatedItem type="host"><titleInfo><title>N Engl J Med</title></titleInfo><part><detail type="volume"><caption>vol.</caption><number>355</number></detail><extent unit="pages"><start>1233</start><end>1243</end></extent></part></relatedItem></relatedItem><relatedItem type="references" displayLabel="trf12876-cit-0022"><titleInfo><title>Soliris® (eculizumab) [package insert]</title></titleInfo><name type="corporate"><namePart>Alexion Pharmaceuticals, Inc.</namePart></name><genre>other</genre></relatedItem><identifier type="istex">9126598165B9466030DCB6AD677E6BA3797C48EF</identifier><identifier type="ark">ark:/67375/WNG-13KSVFTT-T</identifier><identifier type="DOI">10.1111/trf.12876</identifier><identifier type="ArticleID">TRF12876</identifier><accessCondition type="use and reproduction" contentType="copyright">© 2015 AABB© 2014 AABB</accessCondition><recordInfo><recordContentSource authority="ISTEX" authorityURI="https://loaded-corpus.data.istex.fr" valueURI="https://loaded-corpus.data.istex.fr/ark:/67375/XBH-L0C46X92-X">wiley</recordContentSource><recordOrigin>Converted from (version ) to MODS version 3.6.</recordOrigin><recordCreationDate encoding="w3cdtf">2019-11-16</recordCreationDate></recordInfo></mods><json:item><extension>json</extension><original>false</original><mimetype>application/json</mimetype><uri>https://api.istex.fr/ark:/67375/WNG-13KSVFTT-T/record.json</uri></json:item></metadata><serie></serie></istex></record>Pour manipuler ce document sous Unix (Dilib)
EXPLOR_STEP=$WICRI_ROOT/Sante/explor/ChloroquineV1/Data/Istex/Corpus
HfdSelect -h $EXPLOR_STEP/biblio.hfd -nk 001D39 | SxmlIndent | more
Ou
HfdSelect -h $EXPLOR_AREA/Data/Istex/Corpus/biblio.hfd -nk 001D39 | SxmlIndent | more
Pour mettre un lien sur cette page dans le réseau Wicri
{{Explor lien
|wiki= Sante
|area= ChloroquineV1
|flux= Istex
|étape= Corpus
|type= RBID
|clé= ISTEX:9126598165B9466030DCB6AD677E6BA3797C48EF
|texte= Hyperhemolysis syndrome in a patient without a hemoglobinopathy, unresponsive to treatment with eculizumab
}}
| This area was generated with Dilib version V0.6.33. |  |