{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T22:29:22Z","timestamp":1773008962152,"version":"3.50.1"},"reference-count":21,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T00:00:00Z","timestamp":1760054400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T00:00:00Z","timestamp":1760054400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100005881","name":"Hemostasis and Thrombosis Research Society","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100005881","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2025,11]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Neutralising antibodies (inhibitors) against factor VIII can result in severe bleeding in persons with nonsevere haemophilia A (NSHA). The INSIGHT study of 1112 persons with NSHA in a predominantly White population identified 19 different\n                      <jats:italic>F8<\/jats:italic>\n                      missense variants that were associated with inhibitor development.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>\n                      To describe the\n                      <jats:italic>F8<\/jats:italic>\n                      variants and inhibitor development in persons with NSHA in a multiethnic cohort using the\n                      <jats:italic>My Life, Our Future<\/jats:italic>\n                      (MLOF) Research Repository and the American Thrombosis and Hemostasis Network dataset (ATHNdataset).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>The MLOF Research Repository and ATHNdataset were queried for demographic, clinical and genotyping data.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      A total of 1805 persons with NSHA with at least one reportable\n                      <jats:italic>F8<\/jats:italic>\n                      variant and known inhibitor status were included in this study. Inhibitors were developed in 142 (7.9%) persons with NSHA. Inhibitor development occurred in seventy\n                      <jats:italic>F8<\/jats:italic>\n                      variants, of which the majority (\n                      <jats:italic>n<\/jats:italic>\n                      = 67, 95.7%) were missense variants. These 70\u00a0\n                      <jats:italic>F8\u00a0<\/jats:italic>\n                      variants were identified in a total of 1006 (55.7%) persons with NSHA. Race or ethnicity was not associated with inhibitors in persons with NSHA.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      The MLOF Research Repository identified additional\n                      <jats:italic>F8<\/jats:italic>\n                      variants where inhibitor development occurred in a multiethnic cohort of NSHA. Identification of these\n                      <jats:italic>F8<\/jats:italic>\n                      variants can inform both physicians and persons with NSHA to adopt measures to reduce the risk of inhibitor development.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.70143","type":"journal-article","created":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T14:47:03Z","timestamp":1760107623000},"page":"1217-1225","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["<i>F8<\/i>\n                    Variants and Inhibitor Development in a Multiethnic Cohort of Nonsevere Haemophilia A"],"prefix":"10.1111","volume":"31","author":[{"given":"Ming Y.","family":"Lim","sequence":"first","affiliation":[{"name":"Department of Internal Medicine Division of Hematology and Hematologic Malignancies  Salt Lake City Utah USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kristy","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Genetics University of North Carolina  Chapel Hill North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2279-2550","authenticated-orcid":false,"given":"Jill M.","family":"Johnsen","sequence":"additional","affiliation":[{"name":"Department of Medicine University of Washington  Seattle Washington USA"},{"name":"Institute For Stem Cell and Regenerative Medicine University of Washington  Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8930-4304","authenticated-orcid":false,"given":"Nigel S.","family":"Key","sequence":"additional","affiliation":[{"name":"Department of Medicine Division of Hematology and Blood Research Center University of North Carolina At Chapel Hill  Chapel Hill North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,10,10]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365\u20102516.2003.00780.x"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood\u20102013\u201002\u2010483263"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.12990"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102516.2008.01694.x"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood\u20102011\u201009\u2010379453"},{"issue":"3","key":"e_1_2_11_7_1","first-page":"138","article-title":"Mild\/","volume":"14","author":"d'Oiron R.","year":"2008","journal-title":"Haemophilia"},{"issue":"4","key":"e_1_2_11_8_1","first-page":"558","article-title":"Factor VIII","volume":"4","author":"Hay C. R.","year":"1998","journal-title":"Haemophilia"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.15805"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa075760"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.12672"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.14315"},{"key":"e_1_2_11_13_1","unstructured":"European Association for Haemophilia and Allied Disorders (EAHAD) Coagulation Factor VIII Variant Database accessed2024 https:\/\/f8\u2010db.eahad.org."},{"key":"e_1_2_11_14_1","unstructured":"Centers for Disease Control and Prevention (CDC) Hemophilia A Mutation Project (CHAMP) accessed July 1 2024 https:\/\/www.cdc.gov\/hemophilia\/mutation\u2010project."},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1055\/s\u20100029\u20101245111"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1182\/bloodadvances.2020002626"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.26011"},{"key":"e_1_2_11_18_1","unstructured":"Centers for Disease Control and Prevention Community Counts Hemophilia Treatment Center Population Profile accessed July 10 2025 https:\/\/www.cdc.gov\/hemophilia\u2010community\u2010counts\/php\/htc\u2010population\u2010profile\/2023\u2010sept\u2010factor\u2010viii\u2010and\u2010factor\u2010ix.html."},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102516.2011.02739.x"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102516.2011.02509.x"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102516.2011.02700.x"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20102516.2009.02058.x"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70143","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.70143","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70143","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T20:37:58Z","timestamp":1773002278000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.70143"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,10,10]]},"references-count":21,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2025,11]]}},"alternative-id":["10.1111\/hae.70143"],"URL":"https:\/\/doi.org\/10.1111\/hae.70143","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,10,10]]},"assertion":[{"value":"2025-04-16","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-07-02","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-10-10","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}