{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,22]],"date-time":"2026-09-22T18:14:22Z","timestamp":1790100862495,"version":"4.0.1"},"reference-count":39,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2018,6,14]],"date-time":"2018-06-14T00:00:00Z","timestamp":1528934400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"Japan Blood Products Organization"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2018,9]]},"abstract":"<jats:sec><jats:title>Introduction<\/jats:title><jats:p>Immune tolerance induction (<jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content>) was the primary therapeutic approach to eradicate inhibitors in haemophilia patients. Several large <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> registries had been reported, but successful predictors of <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> outcome are still debated. No reports are available on large <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> studies in non\u2010caucasian countries.<\/jats:p><\/jats:sec><jats:sec><jats:title>Aim<\/jats:title><jats:p>We designed a retrospective cohort study of <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> in Japanese haemophilia patients with inhibitor.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Retrospective data were collected from 155 haemophilia (H)A (140 severe\u2010type) and 7 <jats:styled-content style=\"fixed-case\">HB<\/jats:styled-content> (7 severe\u2010type) patients treated at 45 institutions. <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> outcome was centrally reviewed. We defined \u201csuccess\u201d as undetectable inhibitor after 2 consecutive measurements.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> success rate was 71.2% for <jats:styled-content style=\"fixed-case\">HA<\/jats:styled-content> and 83.3% for <jats:styled-content style=\"fixed-case\">HB<\/jats:styled-content>. Cumulated success rates for <jats:styled-content style=\"fixed-case\">HA<\/jats:styled-content> achieving 50% and 75% were 0.7 and 2\u00a0years after treatment, respectively. Significant successful predictors in <jats:styled-content style=\"fixed-case\">HA<\/jats:styled-content> were low\u2010responding inhibitors compared to high\u2010responding inhibitors, shorter time to the start of <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content>, and lower historical and treatment peak titres of inhibitor. Dose regimen (high dose; \u226590 <jats:styled-content style=\"fixed-case\">IU<\/jats:styled-content>\/kg every day, low dose; \u226475 <jats:styled-content style=\"fixed-case\">IU<\/jats:styled-content>\/kg, 3\u00a0d\/wk) and the type of therapeutic product did not affect outcomes. The success rate of salvage <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> using von Willebrand factor\u2010containing factor <jats:styled-content style=\"fixed-case\">VIII<\/jats:styled-content> was 50% (n\u00a0=\u00a06\/12), and patient age at the start of salvage <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> was a significant predictor. The inhibitor recurred in 6 <jats:styled-content style=\"fixed-case\">HA<\/jats:styled-content> cases (3.9%).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The results provided potentially important information for improving future success rates for <jats:styled-content style=\"fixed-case\">ITI<\/jats:styled-content> in inhibitor patients.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.13531","type":"journal-article","created":{"date-parts":[[2018,6,14]],"date-time":"2018-06-14T16:05:18Z","timestamp":1528992318000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["The Japanese Immune Tolerance Induction (J\u2010<scp>ITI<\/scp>) study in haemophilia patients with inhibitor: Outcomes and successful predictors of <scp>ITI<\/scp> 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