{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,11]],"date-time":"2025-11-11T15:43:08Z","timestamp":1762875788349},"reference-count":24,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2016,6,22]],"date-time":"2016-06-22T00:00:00Z","timestamp":1466553600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100004702","name":"Baxter International","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004702","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004326","name":"Bayer","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004326","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100006314","name":"Biogen Idec","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006314","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004337","name":"Roche","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004337","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100008322","name":"CSL Behring","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100008322","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004191","name":"Novo Nordisk","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004191","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004319","name":"Pfizer","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004319","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2016,11]]},"abstract":"<jats:sec><jats:title>Introduction<\/jats:title><jats:p>Immune tolerance induction (ITI) is a standard intervention to eradicate inhibitors in haemophilic patients. However, information on the long\u2010term condition of patients who eradicated the inhibitor totally or partially after ITI is scarce.<\/jats:p><\/jats:sec><jats:sec><jats:title>Aim<\/jats:title><jats:p>To perform a long\u2010term follow\u2010up to describe the status of patients reported as ITI success in the G\u2010ITI study.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>This was an international, multicentre, observational, retrospective study of the 57 haemophilic patients who were reported as ITI success in the G\u2010ITI study. Demographics and post\u2010ITI clinical data recorded until January 2015 were extracted from the medical records. A descriptive analysis was undertaken.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Forty\u2010four patients were evaluable. Post\u2010ITI follow\u2010up was 9.1 years in average. Thirty\u2010seven target joints were affected in 21 patients; 38 patients presented bleeding with a mean of 1.0 \u00b1 1.2 episodes year<jats:sup>\u22121<\/jats:sup>, most of them (271\/330) treated with Fanhdi<jats:sup>\u00ae<\/jats:sup> (Grifols). Around half of the patients underwent at least one surgical procedure. Most venous access complications were of expected nature, requiring hospital stay in practically all cases. Fanhdi was used in 42 patients as the regular haemophilia treatment after ITI, mainly prophylactically. Three patients (6.8%) who were being treated with Fanhdi (prophylaxis), Kogenate (prophylaxis) and Emoclot (on demand), respectively, showed inhibitor relapse (at 29, 53 and 13 months after ITI, with 0.9, 3.65 and 12.5 BU respectively). All of them were successfully tolerized after rescue ITI.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>After ITI success, all patients continued with regular successful FVIII treatment for haemophilia for more than 9 years. The few inhibitor relapses were successfully overcome after rescue ITI.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.12986","type":"journal-article","created":{"date-parts":[[2016,6,23]],"date-time":"2016-06-23T05:59:01Z","timestamp":1466661541000},"page":"859-865","source":"Crossref","is-referenced-by-count":19,"title":["Long\u2010term outcome of haemophilia A patients after successful immune tolerance induction therapy using a single plasma\u2010derived FVIII\/VWF product: the long\u2010term ITI study"],"prefix":"10.1111","volume":"22","author":[{"given":"V.","family":"Jim\u00e9nez\u2010Yuste","sequence":"first","affiliation":[{"name":"Haematology Department Hospital Universitario La Paz Universidad Aut\u00f3noma de Madrid Madrid Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"J.","family":"Oldenburg","sequence":"additional","affiliation":[{"name":"University Clinic Bonn Institute of Experimental Haematology and Transfusion Medicine Bonn Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"S.","family":"Rangarajan","sequence":"additional","affiliation":[{"name":"Haemophilia, Haemostasis &amp; Thrombosis Centre Hampshire Hospitals NHS Foundation Trust Basingstoke UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"R.","family":"Peir\u00f3\u2010Jord\u00e1n","sequence":"additional","affiliation":[{"name":"Scientific and Medical Affairs Department Grifols Sant Cugat del Vall\u00e8s Barcelona Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"E.","family":"Santagostino","sequence":"additional","affiliation":[{"name":"Centro Emofilia e Trombosi \u201cAngelo Bianchi Bonomi\u201d Fondazione IRCCS Ca' Granda Milano ltalia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2016,6,22]]},"reference":[{"key":"e_1_2_7_2_1","first-page":"1548","volume-title":"Hematology of Infancy and Childhood","author":"Montgomery RR","year":"2003"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/0140-6736(92)90874-3"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.12942"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2010-09-308668"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1423-0410.1996.tb01346.x"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood.V86.3.983.983"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198804143181503"},{"issue":"2","key":"e_1_2_7_9_1","first-page":"1081","article-title":"Immune tolerance induction in adult severe haemophilia A patients with a single FVIII\/VWF product: the UK experience","volume":"11","author":"Rangarajan S","year":"2013","journal-title":"J Thromb Haemost"},{"key":"e_1_2_7_10_1","first-page":"EREP05","article-title":"The role of plasma\u2010derived factor VIII\/von Willebrand factor concentrates in the treatment of hemophilia A patients","volume":"88","author":"Auerswald G","year":"2003","journal-title":"Haematologica"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.mbc.0000167660.30886.e9"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12774"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.thromres.2014.09.010"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.2003.00781.x"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12263"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12521"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2006.01312.x"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.2106\/00004623-197759030-00001"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12375"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12439"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12563"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2007.01497.x"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12051"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2011-08-369132"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.13143"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fhae.12986","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fhae.12986","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.12986","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.12986","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.12986","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,23]],"date-time":"2023-09-23T20:26:07Z","timestamp":1695500767000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.12986"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,6,22]]},"references-count":24,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2016,11]]}},"alternative-id":["10.1111\/hae.12986"],"URL":"https:\/\/doi.org\/10.1111\/hae.12986","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,6,22]]}}}