{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,26]],"date-time":"2026-07-26T23:14:59Z","timestamp":1785107699705,"version":"3.55.0"},"reference-count":35,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2013,11,19]],"date-time":"2013-11-19T00:00:00Z","timestamp":1384819200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2014,1]]},"abstract":"Summary<\/jats:title>Most studies on immune tolerance induction (ITI<\/jats:styled-content>) therapy in haemophilia A patients are focused on primary ITI<\/jats:styled-content> in children. Here we report on the ITI<\/jats:styled-content> outcome in a large retrospective cohort, including adults and patients with rescue ITI<\/jats:styled-content>, treated with a pdFVIII<\/jats:styled-content>\/VWF<\/jats:styled-content> concentrate. Retrospective data from haemophilic patients (FVIII<\/jats:styled-content>< 2%) with inhibitors from 22 centres in Spain, Italy and Germany, who underwent primary or rescue ITI<\/jats:styled-content> with pdFVIII<\/jats:styled-content>\/VWF<\/jats:styled-content> concentrate, were collected. Complete success (CS<\/jats:styled-content>), partial success (PS<\/jats:styled-content>) and failure were defined based on the criteria of the consensus recommendations of the 2006 International ITI<\/jats:styled-content> Workshop. A total of 41 cases of primary ITI<\/jats:styled-content> (32 children and 9 adults) and 19 cases of rescue ITI<\/jats:styled-content> (17 children and 2 adults) were evaluated. Success (CS<\/jats:styled-content>+PS<\/jats:styled-content>) rate of 87% was achieved in primary ITI<\/jats:styled-content> and 74% in the higher risk profile of rescue ITI<\/jats:styled-content>. Eight of nine (85%) patients with poorest prognosis (three or more of the known risk factors of poor response to ITI<\/jats:styled-content>) achieved success (CS<\/jats:styled-content>+PS<\/jats:styled-content>). CS<\/jats:styled-content> of 100% was observed in eight primary ITI<\/jats:styled-content> patients with titre at start of ITI<\/jats:styled-content> \u22642.5\u00a0BU and inhibitor peak \u226425\u00a0BU. The favourable response rates in primary and rescue ITI<\/jats:styled-content> in children and in adult patients, even in the presence of poor prognostic factors, should be encouraged for broadening the indication of immune tolerance therapy in haemophilia A patients with inhibitors.<\/jats:p>","DOI":"10.1111\/hae.12263","type":"journal-article","created":{"date-parts":[[2013,11,20]],"date-time":"2013-11-20T04:26:56Z","timestamp":1384921616000},"page":"83-91","source":"Crossref","is-referenced-by-count":56,"title":["Primary and rescue immune tolerance induction in children and adults: a multicentre international study with a VWF<\/scp>\u2010containing plasma\u2010derived FVIII<\/scp> concentrate"],"prefix":"10.1111","volume":"20","author":[{"given":"J.","family":"Oldenburg","sequence":"first","affiliation":[{"name":"Institute of Experimental Haematology and Transfusion Medicine University Clinic Bonn Bonn Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"V.","family":"Jim\u00e9nez\u2010Yuste","sequence":"additional","affiliation":[{"name":"Hematology Department Hospital Universitario La Paz Universidad Aut\u00f3noma de Madrid Madrid Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R.","family":"Peir\u00f3\u2010Jord\u00e1n","sequence":"additional","affiliation":[{"name":"Scientific and Medical Affairs Grifols S.A. Barcelona Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L. M.","family":"Aledort","sequence":"additional","affiliation":[{"name":"Department of Medicine Mount Sinai Medical Center New York NY USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"E.","family":"Santagostino","sequence":"additional","affiliation":[{"name":"Angelo Bianchi Bonomi Hemophilia and Thrombosis Center IRCCS C\u00e0 Granda Foundation Ospedale Maggiore Policlinico Milan Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2013,11,19]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.7326\/0003-4819-114-8-641"},{"key":"e_1_2_8_3_1","first-page":"11","article-title":"Viral safety evaluation of plasma\u2010derived therapeutic products","volume":"118","author":"Farshid M","year":"2004","journal-title":"Dev Biol (Basel)"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2012-01-394411"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/0140-6736(93)91593-B"},{"key":"e_1_2_8_6_1","first-page":"EREP04","article-title":"Epidemiology of inhibitors and current treatment strategies","volume":"88","author":"Kreuz W","year":"2003","journal-title":"Haematologica"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1538-7836.2004.00759.x"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2006-04-017988"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1097\/00001721-200004001-00009"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2003-03-0941"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1423-0410.1996.tb01346.x"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2141.2010.08263.x"},{"key":"e_1_2_8_13_1","first-page":"1","article-title":"Immunotolerance induction treatments in hemophilia","volume":"1","author":"Haya S","year":"2009","journal-title":"J Coagul Disord"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1159\/000056713"},{"key":"e_1_2_8_15_1","first-page":"62","article-title":"Immunetolerance as treatment of alloantibodies to factor VIII in hemophilia. 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