{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,25]],"date-time":"2026-02-25T16:05:36Z","timestamp":1772035536786,"version":"3.50.1"},"reference-count":37,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2019,5,21]],"date-time":"2019-05-21T00:00:00Z","timestamp":1558396800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000030","name":"Centers for Disease Control and Prevention","doi-asserted-by":"publisher","award":["U27\/CCU812106"],"award-info":[{"award-number":["U27\/CCU812106"]}],"id":[{"id":"10.13039\/100000030","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100006010","name":"Bayer Schering","doi-asserted-by":"publisher","award":["Beth Boulden Warren - Bayer Hemophilia Awards Prog"],"award-info":[{"award-number":["Beth Boulden Warren - Bayer Hemophilia Awards Prog"]}],"id":[{"id":"10.13039\/501100006010","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100006010","name":"Bayer Schering","doi-asserted-by":"publisher","award":["Donation of Kogenate for study"],"award-info":[{"award-number":["Donation of Kogenate for study"]}],"id":[{"id":"10.13039\/501100006010","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100005881","name":"Hemostasis and Thrombosis Research Society","doi-asserted-by":"publisher","award":["Beth Boulden Warren - HTRS\/Novo Nordisk Clinical F"],"award-info":[{"award-number":["Beth Boulden Warren - HTRS\/Novo Nordisk Clinical F"]}],"id":[{"id":"10.13039\/100005881","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007305","name":"Maternal and Child Health Bureau","doi-asserted-by":"publisher","award":["2H30MC24049"],"award-info":[{"award-number":["2H30MC24049"]}],"id":[{"id":"10.13039\/100007305","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000098","name":"NIH Clinical Center","doi-asserted-by":"publisher","award":["R00069"],"award-info":[{"award-number":["R00069"]}],"id":[{"id":"10.13039\/100000098","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2019,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>The Joint Outcome Study (JOS) demonstrated that previously untreated children with severe haemophilia A treated with prophylactic factor VIII (FVIII) concentrate had superior joint outcomes at age 6\u00a0years compared to those children treated episodically for bleeding. However, variation in joint outcome within each treatment arm was not well explained.<\/jats:p><\/jats:sec><jats:sec><jats:title>Aim<\/jats:title><jats:p>In this study, we sought to better understand variation in joint outcomes at age 6\u00a0years in participants of the JOS.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We evaluated the influence of FVIII half\u2010life, treatment adherence, constitutional coagulant and anticoagulant proteins, and global assays on joint outcomes (number of joint bleeds, total number of bleeds, total MRI score and joint physical exam score). Logistic regression was used to evaluate the association of variables with joint failure status on MRI, defined as presence of subchondral cyst, surface erosion or joint\u2010space narrowing. Each parameter was also correlated with each joint outcome using Spearman correlations.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Prophylaxis treatment arm and FVIII trough were each found to reduce risk of joint failure on univariate logistic regression analysis. When controlling for treatment arm, FVIII trough was no longer significant, likely because of the high level of covariation between these variables. We found no consistent correlation between any laboratory assay performed and any joint outcome parameter measured.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>In the JOS, the effect of prescribed prophylactic FVIII infusions on joint outcome overshadowed the contribution of treatment adherence, FVIII half\u2010life, global assays of coagulation and constitutional coagulation proteins. (ClinicalTrials.gov number, NCT00207597).<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.13778","type":"journal-article","created":{"date-parts":[[2019,5,22]],"date-time":"2019-05-22T03:38:24Z","timestamp":1558496304000},"page":"867-875","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Factor VIII prophylaxis effects outweigh other hemostasis contributors in predicting severe haemophilia A joint outcomes"],"prefix":"10.1111","volume":"25","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4416-3031","authenticated-orcid":false,"given":"Beth Boulden","family":"Warren","sequence":"first","affiliation":[{"name":"University of Colorado Anschutz Medical Campus Aurora Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Linda","family":"Jacobson","sequence":"additional","affiliation":[{"name":"University of Colorado Anschutz Medical Campus Aurora Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2391-2016","authenticated-orcid":false,"given":"Christine","family":"Kempton","sequence":"additional","affiliation":[{"name":"Emory University School of Medicine Atlanta Georgia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"George R.","family":"Buchanan","sequence":"additional","affiliation":[{"name":"Children's Medical Center at Dallas University of Texas Southwestern Medical Center Dallas Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael","family":"Recht","sequence":"additional","affiliation":[{"name":"Phoenix Children's Hospital Phoenix Arizona"},{"name":"Oregon Health &amp; Science University Portland Oregon"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Deborah","family":"Brown","sequence":"additional","affiliation":[{"name":"University of Texas Houston Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Cindy","family":"Leissinger","sequence":"additional","affiliation":[{"name":"Tulane University New Orleans Louisiana"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amy D.","family":"Shapiro","sequence":"additional","affiliation":[{"name":"Indiana Hemophilia and Thrombosis Center Indianapolis Indiana"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Thomas C.","family":"Abshire","sequence":"additional","affiliation":[{"name":"Emory University School of Medicine Atlanta Georgia"},{"name":"Blood Research Institute BloodCenter of Wisconsin\/Versiti Milwaukee Wisconsin"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5215-1482","authenticated-orcid":false,"given":"Marilyn J.","family":"Manco\u2010Johnson","sequence":"additional","affiliation":[{"name":"University of Colorado Anschutz Medical Campus Aurora Colorado"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"name":"for the Joint Outcome Study Group Investigators","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2019,5,21]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa067659"},{"key":"e_1_2_10_3_1","unstructured":"HemophiliaN.Foundation Medical and Scientific Advisory Council (MASAC). 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