{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,7]],"date-time":"2026-10-07T09:51:18Z","timestamp":1791366678971,"version":"4.3.1"},"reference-count":29,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2026,7,23]],"date-time":"2026-07-23T00:00:00Z","timestamp":1784764800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,7,23]],"date-time":"2026-07-23T00:00:00Z","timestamp":1784764800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/501100004359","name":"Vetenskapsr\u00e5det","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004359","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100011077","name":"Sk\u00e5nes universitetssjukhus","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100011077","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["European J of Haematology"],"published-print":{"date-parts":[[2026,11]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Immune tolerance induction (ITI) to eradicate an immune response against factor VIII (FVIII) has been used in hemophilia A (HA) with various success rates, costs, and treatment burden. The present study aimed to identify markers predictive of ITI success. Data were accrued from 237 patients with severe HA (FVIII level &lt;\u20091%), history of persistent FVIII inhibitors \u2265\u20091 BU\/mL and having performed ITI. Constitutional and clinical variables associated with ITI outcome were evaluated and multivariable logistic regression models developed. The\n                    <jats:italic>F8<\/jats:italic>\n                    variant, age at inhibitor detection, and historical peak inhibitor titer were associated with ITI outcome. HLA DRB1*1501 and DQB1*0602 were linked to each other, but not associated with ITI outcome. In a multivariable\u2010adjusted model, large gene deletions, peak titer \u2265\u200925\u2009BU\/mL, and higher age at inhibitor detection were associated with a significantly lower ITI success rate. The discriminatory power of a predictive model was 0.87 (95% confidence interval [CI], 0.85\u20130.93) with the historical peak titer and 0.69 (95% CI, 0.64\u20130.80) without. We conclude that the peak inhibitor titer is a key determinant for ITI outcome and additional constitutional markers need to be defined for useful prediction in the clinical setting.\n                  <\/jats:p>","DOI":"10.1111\/ejh.70269","type":"journal-article","created":{"date-parts":[[2026,7,23]],"date-time":"2026-07-23T23:22:11Z","timestamp":1784848931000},"page":"1292-1300","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Determinants of Immune Tolerance Induction Outcomes in 237 Persons With Severe Hemophilia A"],"prefix":"10.1111","volume":"117","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8651-101X","authenticated-orcid":false,"given":"Marcus","family":"Fager Ferrari","sequence":"first","affiliation":[{"name":"Department of Hematology, Oncology and Radiation Physics Sk\u00e5ne University Hospital  Malm\u00f6 Sweden"},{"name":"Department of Translational Medicine Lund University  Malm\u00f6 Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6481-0895","authenticated-orcid":false,"given":"Josefin","family":"Roslund","sequence":"additional","affiliation":[{"name":"Department of Hematology, Oncology and Radiation Physics Sk\u00e5ne University Hospital  Malm\u00f6 Sweden"},{"name":"Department of Translational Medicine Lund University  Malm\u00f6 Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eric","family":"Manderstedt","sequence":"additional","affiliation":[{"name":"Center for Primary Health Care Research, Department of Clinical Sciences Lund University  Malm\u00f6 Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1337-7195","authenticated-orcid":false,"given":"Erik","family":"Berntorp","sequence":"additional","affiliation":[{"name":"Department of Translational Medicine Lund University  Malm\u00f6 Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8500-2483","authenticated-orcid":false,"given":"Jan","family":"Astermark","sequence":"additional","affiliation":[{"name":"Department of Hematology, Oncology and Radiation Physics Sk\u00e5ne University Hospital  Malm\u00f6 Sweden"},{"name":"Department of Translational Medicine Lund University  Malm\u00f6 Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"Malm\u00f6 and Hemophilia Inhibitor Genetics Study Investigators","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,7,23]]},"reference":[{"key":"e_1_2_12_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(11)61139-2"},{"key":"e_1_2_12_3_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2014-08-535328"},{"key":"e_1_2_12_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.12990"},{"key":"e_1_2_12_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13762"},{"key":"e_1_2_12_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14381"},{"key":"e_1_2_12_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14470"},{"key":"e_1_2_12_8_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2011-08-369132"},{"key":"e_1_2_12_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13445"},{"key":"e_1_2_12_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.rpth.2022.100020"},{"key":"e_1_2_12_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(24)02139-1"},{"key":"e_1_2_12_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ymthe.2025.03.043"},{"key":"e_1_2_12_13_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0038-1655945"},{"key":"e_1_2_12_14_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0038-1655944"},{"key":"e_1_2_12_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12168"},{"key":"e_1_2_12_16_1","doi-asserted-by":"publisher","DOI":"10.3389\/fmed.2022.903838"},{"key":"e_1_2_12_17_1","doi-asserted-by":"publisher","DOI":"10.3389\/fgene.2025.1506862"},{"key":"e_1_2_12_18_1","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.2022.281279"},{"issue":"1","key":"e_1_2_12_19_1","first-page":"14","article-title":"A Novel Genetic Classification of Inhibitor Risk for F8 Nonsense Mutations Based on Immunogenic Profiling of Ribosomal Readthrough in EAHAD Database","volume":"32","author":"Testa M. 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