Late immune tolerance induction in haemophilia A patients

Summary The incidence of inhibitor development in patients with severe haemophilia A is approximately 30%. Immune tolerance induction (ITI) is commonly utilized to eradicate these antibodies and is successful in 63–100% of cases. Potential predictors of a poor outcome in ITI include a high preinduct...

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Published in:Haemophilia : the official journal of the World Federation of Hemophilia 2013-05, Vol.19 (3), p.445-448
Main Authors: Meeks, S. L., Chapman, R. L., Kempton, C., Dunn, A. L.
Format: Article
Language:English
Subjects:
Adolescent
Adult
Age Factors
Child
Child, Preschool
Cohort Studies
factor VIII
Factor VIII - antagonists & inhibitors
Factor VIII - immunology
Factor VIII - therapeutic use
haemophilia A
Hemophilia A - drug therapy
Hemophilia A - immunology
Humans
Immune Tolerance
inhibitor
Isoantibodies - blood
Male
Young Adult
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cited_by cdi_FETCH-LOGICAL-c3967-b0f3bfd001486a3888debee5fb1642cea4fe4e5ea45e8037348f69fcb0642bb53
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container_issue 3
container_start_page 445
container_title Haemophilia : the official journal of the World Federation of Hemophilia
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creator Meeks, S. L.
Chapman, R. L.
Kempton, C.
Dunn, A. L.
description Summary The incidence of inhibitor development in patients with severe haemophilia A is approximately 30%. Immune tolerance induction (ITI) is commonly utilized to eradicate these antibodies and is successful in 63–100% of cases. Potential predictors of a poor outcome in ITI include a high preinduction titre, high historical peak titre, older age at start of ITI and prolonged interval from diagnosis to start of ITI. The goal of this study was to characterize the outcomes of patients from our centre who have undergone late ITI, many of whom had poor prognostic features. Medical records of patients in our centre with severe/moderately severe haemophilia A (6 h). Three patients are partially tolerized (have low responding inhibitor, variable FVIII recovery and successfully treated with FVIII products). Two patients are not tolerized. Some patients with haemophilia A and long‐standing inhibitors may benefit from ITI.
doi_str_mv 10.1111/hae.12077
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Nine patients underwent late ITI between January 1999 and December 2011. Within this cohort, 7 (78%) patients were black, 6 (67%) were &lt;21 years old and 4 (44%) had a family history of inhibitor. Three patients had previously received ITI unsuccessfully. To date, 4 (44%) patients are tolerized (persistently negative inhibitor titre, FVIII recovery &gt;66% and successfully treated with FVIII products ±FVIII t½ of &gt;6 h). Three patients are partially tolerized (have low responding inhibitor, variable FVIII recovery and successfully treated with FVIII products). Two patients are not tolerized. 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L.</creatorcontrib><creatorcontrib>Chapman, R. L.</creatorcontrib><creatorcontrib>Kempton, C.</creatorcontrib><creatorcontrib>Dunn, A. L.</creatorcontrib><title>Late immune tolerance induction in haemophilia A patients</title><title>Haemophilia : the official journal of the World Federation of Hemophilia</title><addtitle>Haemophilia</addtitle><description>Summary The incidence of inhibitor development in patients with severe haemophilia A is approximately 30%. Immune tolerance induction (ITI) is commonly utilized to eradicate these antibodies and is successful in 63–100% of cases. Potential predictors of a poor outcome in ITI include a high preinduction titre, high historical peak titre, older age at start of ITI and prolonged interval from diagnosis to start of ITI. The goal of this study was to characterize the outcomes of patients from our centre who have undergone late ITI, many of whom had poor prognostic features. Medical records of patients in our centre with severe/moderately severe haemophilia A (&lt;2% FVIII activity) and history of inhibitor were reviewed. Data were ed from all patients who attempted late ITI. Nine patients underwent late ITI between January 1999 and December 2011. Within this cohort, 7 (78%) patients were black, 6 (67%) were &lt;21 years old and 4 (44%) had a family history of inhibitor. Three patients had previously received ITI unsuccessfully. To date, 4 (44%) patients are tolerized (persistently negative inhibitor titre, FVIII recovery &gt;66% and successfully treated with FVIII products ±FVIII t½ of &gt;6 h). Three patients are partially tolerized (have low responding inhibitor, variable FVIII recovery and successfully treated with FVIII products). Two patients are not tolerized. Some patients with haemophilia A and long‐standing inhibitors may benefit from ITI.</description><subject>Adolescent</subject><subject>Adult</subject><subject>Age Factors</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Cohort Studies</subject><subject>factor VIII</subject><subject>Factor VIII - antagonists &amp; inhibitors</subject><subject>Factor VIII - immunology</subject><subject>Factor VIII - therapeutic use</subject><subject>haemophilia A</subject><subject>Hemophilia A - drug therapy</subject><subject>Hemophilia A - immunology</subject><subject>Humans</subject><subject>Immune Tolerance</subject><subject>inhibitor</subject><subject>Isoantibodies - blood</subject><subject>Male</subject><subject>Young Adult</subject><issn>1351-8216</issn><issn>1365-2516</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2013</creationdate><recordtype>article</recordtype><recordid>eNqNkMtOwzAQRS0EoqWw4AdQlrBIa2dix1m2VR-gCoQEQmJjOelENeRR4kTQv8elpTskvJmRfeZYcwm5ZLTP3BmsNPZZQKPoiHQZCO4HnInjbc-ZLwMmOuTM2jdKGQRUnJJOAEEcUgFdEi90g54pirZEr6lyrHWZuoty2aaNqUrXeU5fVOuVyY32ht5aNwbLxp6Tk0znFi_2tUeep5On8dxfPMxux8OFn0IsIj-hGSTZ0v0dSqFBSrnEBJFnCRNhkKIOMwyRu8pRUogglJmIszSh7jlJOPTI9c67rquPFm2jCmNTzHNdYtVa5RaOQsmB_wcFCTEEwBx6s0PTurK2xkyta1PoeqMYVdtQldta_YTq2Ku9tk0KXB7I3xQdMNgBnybHzd8mNR9OfpX-bsLYBr8OE7p-VyKCiKuX-5m6e4z4iL9O1Qi-ATLejmw</recordid><startdate>201305</startdate><enddate>201305</enddate><creator>Meeks, S. L.</creator><creator>Chapman, R. L.</creator><creator>Kempton, C.</creator><creator>Dunn, A. L.</creator><general>Blackwell Publishing Ltd</general><scope>BSCLL</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>7T5</scope><scope>H94</scope></search><sort><creationdate>201305</creationdate><title>Late immune tolerance induction in haemophilia A patients</title><author>Meeks, S. L. ; Chapman, R. L. ; Kempton, C. ; Dunn, A. 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L.</creatorcontrib><creatorcontrib>Chapman, R. L.</creatorcontrib><creatorcontrib>Kempton, C.</creatorcontrib><creatorcontrib>Dunn, A. L.</creatorcontrib><collection>Istex</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>Immunology Abstracts</collection><collection>AIDS and Cancer Research Abstracts</collection><jtitle>Haemophilia : the official journal of the World Federation of Hemophilia</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Meeks, S. L.</au><au>Chapman, R. L.</au><au>Kempton, C.</au><au>Dunn, A. L.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Late immune tolerance induction in haemophilia A patients</atitle><jtitle>Haemophilia : the official journal of the World Federation of Hemophilia</jtitle><addtitle>Haemophilia</addtitle><date>2013-05</date><risdate>2013</risdate><volume>19</volume><issue>3</issue><spage>445</spage><epage>448</epage><pages>445-448</pages><issn>1351-8216</issn><eissn>1365-2516</eissn><notes>ArticleID:HAE12077</notes><notes>ark:/67375/WNG-JQ75B5ZF-B</notes><notes>istex:ED56727C04FD058C084021C4226C181CAB615677</notes><notes>ObjectType-Case Study-2</notes><notes>SourceType-Scholarly Journals-1</notes><notes>ObjectType-Feature-4</notes><notes>content type line 23</notes><notes>ObjectType-Report-1</notes><notes>ObjectType-Article-3</notes><notes>ObjectType-Article-2</notes><notes>ObjectType-Feature-1</notes><abstract>Summary The incidence of inhibitor development in patients with severe haemophilia A is approximately 30%. Immune tolerance induction (ITI) is commonly utilized to eradicate these antibodies and is successful in 63–100% of cases. Potential predictors of a poor outcome in ITI include a high preinduction titre, high historical peak titre, older age at start of ITI and prolonged interval from diagnosis to start of ITI. The goal of this study was to characterize the outcomes of patients from our centre who have undergone late ITI, many of whom had poor prognostic features. Medical records of patients in our centre with severe/moderately severe haemophilia A (&lt;2% FVIII activity) and history of inhibitor were reviewed. Data were ed from all patients who attempted late ITI. Nine patients underwent late ITI between January 1999 and December 2011. Within this cohort, 7 (78%) patients were black, 6 (67%) were &lt;21 years old and 4 (44%) had a family history of inhibitor. Three patients had previously received ITI unsuccessfully. To date, 4 (44%) patients are tolerized (persistently negative inhibitor titre, FVIII recovery &gt;66% and successfully treated with FVIII products ±FVIII t½ of &gt;6 h). Three patients are partially tolerized (have low responding inhibitor, variable FVIII recovery and successfully treated with FVIII products). Two patients are not tolerized. Some patients with haemophilia A and long‐standing inhibitors may benefit from ITI.</abstract><cop>England</cop><pub>Blackwell Publishing Ltd</pub><pmid>23294063</pmid><doi>10.1111/hae.12077</doi><tpages>4</tpages></addata></record>
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source Wiley Online Library
subjects Adolescent
Adult
Age Factors
Child
Child, Preschool
Cohort Studies
factor VIII
Factor VIII - antagonists & inhibitors
Factor VIII - immunology
Factor VIII - therapeutic use
haemophilia A
Hemophilia A - drug therapy
Hemophilia A - immunology
Humans
Immune Tolerance
inhibitor
Isoantibodies - blood
Male
Young Adult
title Late immune tolerance induction in haemophilia A patients
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