{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,3]],"date-time":"2026-11-03T02:36:25Z","timestamp":1793673385758,"version":"4.5.1"},"reference-count":26,"publisher":"Elsevier BV","issue":"1","license":[{"start":{"date-parts":[[2024,1,1]],"date-time":"2024-01-01T00:00:00Z","timestamp":1704067200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2024,1,1]],"date-time":"2024-01-01T00:00:00Z","timestamp":1704067200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2025,1,1]],"date-time":"2025-01-01T00:00:00Z","timestamp":1735689600000},"content-version":"vor","delay-in-days":366,"URL":"http:\/\/www.elsevier.com\/open-access\/userlicense\/1.0\/"}],"funder":[{"DOI":"10.13039\/501100022776","name":"Stichting Haemophilia","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100022776","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002999","name":"Ministerie van Volksgezondheid, Welzijn en Sport","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100002999","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["jthjournal.org","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Journal of Thrombosis and Haemostasis"],"published-print":{"date-parts":[[2024,1]]},"DOI":"10.1016\/j.jtha.2023.09.029","type":"journal-article","created":{"date-parts":[[2023,10,7]],"date-time":"2023-10-07T11:36:51Z","timestamp":1696678611000},"page":"152-162","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":21,"title":["Patients with moderate hemophilia A and B with a severe bleeding phenotype have an increased burden of disease"],"prefix":"10.1016","volume":"22","author":[{"given":"Marieke J.A.","family":"Verhagen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erna C.","family":"van Balen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicole M.A.","family":"Blijlevens","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michiel","family":"Coppens","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Waander L.","family":"van Heerde","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank W.G.","family":"Leebeek","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sanna R.","family":"Rijpma","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lize F.D.","family":"van Vulpen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Samantha C.","family":"Gouw","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saskia E.M.","family":"Schols","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.jtha.2023.09.029_bib1","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1111\/hae.14046","article-title":"WFH Guidelines for the Management of Hemophilia, 3rd edition","volume":"26","author":"Srivastava","year":"2020","journal-title":"Haemophilia"},{"key":"10.1016\/j.jtha.2023.09.029_bib2","doi-asserted-by":"crossref","first-page":"128","DOI":"10.1055\/s-2008-1066024","article-title":"The phenotypic heterogeneity of severe hemophilia","volume":"34","author":"Jayandharan","year":"2008","journal-title":"Semin Thromb Hemost"},{"key":"10.1016\/j.jtha.2023.09.029_bib3","doi-asserted-by":"crossref","first-page":"3","DOI":"10.3109\/ort.1965.36.suppl-77.01","article-title":"Haemophilia in Sweden. VII. Incidence, treatment and prophylaxis of arthropathy and other musculo-skeletal manifestations of haemophilia A and B","author":"Ahlberg","year":"1965","journal-title":"Acta Orthop Scand Suppl"},{"key":"10.1016\/j.jtha.2023.09.029_bib4","first-page":"s330","article-title":"Outcome in moderate haemophilia","volume":"12","author":"den Uijl","year":"2014","journal-title":"Blood Transfus"},{"key":"10.1016\/j.jtha.2023.09.029_bib5","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1111\/j.1365-2516.2008.01837.x","article-title":"Clinical outcome of moderate haemophilia compared with severe and mild haemophilia","volume":"15","author":"den Uijl","year":"2009","journal-title":"Haemophilia"},{"key":"10.1016\/j.jtha.2023.09.029_bib6","doi-asserted-by":"crossref","first-page":"849","DOI":"10.1111\/j.1365-2516.2011.02539.x","article-title":"Clinical severity of haemophilia A: does the classification of the 1950s still stand?","volume":"17","author":"Den Uijl","year":"2011","journal-title":"Haemophilia"},{"key":"10.1016\/j.jtha.2023.09.029_bib7","doi-asserted-by":"crossref","first-page":"2394","DOI":"10.1111\/jth.15424","article-title":"Health and treatment outcomes of patients with hemophilia in the Netherlands, 1972-2019","volume":"19","author":"Hassan","year":"2021","journal-title":"J\u00a0Thromb Haemost"},{"key":"10.1016\/j.jtha.2023.09.029_bib8","doi-asserted-by":"crossref","first-page":"426","DOI":"10.1111\/hae.12326","article-title":"Joint health scores in a haemophilia A cohort from Pakistan with minimal or no access to factor VIII concentrate: correlation with thrombin generation and underlying mutation","volume":"20","author":"Khanum","year":"2014","journal-title":"Haemophilia"},{"key":"10.1016\/j.jtha.2023.09.029_bib9","doi-asserted-by":"crossref","first-page":"2494","DOI":"10.1111\/jth.12025","article-title":"Magnetic resonance imaging and joint outcomes in boys with severe hemophilia A treated with tailored primary prophylaxis in Canada","volume":"10","author":"Kraft","year":"2012","journal-title":"J\u00a0Thromb Haemost"},{"key":"10.1016\/j.jtha.2023.09.029_bib10","doi-asserted-by":"crossref","first-page":"205","DOI":"10.1111\/hae.13616","article-title":"Treatment regimens and outcomes in severe and moderate haemophilia A in the UK: The THUNDER study","volume":"25","author":"Scott","year":"2019","journal-title":"Haemophilia"},{"key":"10.1016\/j.jtha.2023.09.029_bib11","doi-asserted-by":"crossref","first-page":"1126","DOI":"10.1111\/jth.15676","article-title":"Joint status of patients with nonsevere hemophilia A","volume":"20","author":"Zwagemaker","year":"2022","journal-title":"J\u00a0Thromb Haemost"},{"key":"10.1016\/j.jtha.2023.09.029_bib12","doi-asserted-by":"crossref","first-page":"723","DOI":"10.1055\/s-0033-1354422","article-title":"Arthropathy in patients with moderate hemophilia a: a systematic review of the literature","volume":"39","author":"Di Minno","year":"2013","journal-title":"Semin Thromb Hemost"},{"key":"10.1016\/j.jtha.2023.09.029_bib13","doi-asserted-by":"crossref","first-page":"619","DOI":"10.1111\/hae.13493","article-title":"Individual thrombin generation and spontaneous bleeding rate during personalized prophylaxis with Nuwiq(\u00ae) (human-cl rhFVIII) in previously treated patients with severe haemophilia A","volume":"24","author":"Dargaud","year":"2018","journal-title":"Haemophilia"},{"key":"10.1016\/j.jtha.2023.09.029_bib14","article-title":"In patients with hemophilia, a decreased thrombin generation profile is associated with a severe bleeding phenotype","volume":"7","author":"Verhagen","year":"2023","journal-title":"Res Pract Thromb Haemost"},{"key":"10.1016\/j.jtha.2023.09.029_bib15","doi-asserted-by":"crossref","first-page":"3422","DOI":"10.1182\/bloodadvances.2021006403","article-title":"Long-term efficacy and safety of subcutaneous concizumab prophylaxis in hemophilia A and hemophilia A\/B with inhibitors","volume":"6","author":"Shapiro","year":"2022","journal-title":"Blood Adv"},{"key":"10.1016\/j.jtha.2023.09.029_bib16","doi-asserted-by":"crossref","first-page":"1018","DOI":"10.1056\/NEJMoa2002699","article-title":"BIVV001 fusion protein as factor VIII replacement therapy for hemophilia A","volume":"383","author":"Konkle","year":"2020","journal-title":"N\u00a0Engl J Med"},{"key":"10.1016\/j.jtha.2023.09.029_bib17","doi-asserted-by":"crossref","first-page":"2687","DOI":"10.1111\/jth.15454","article-title":"Validation of PROMIS profile-29 in adults with hemophilia in the Netherlands","volume":"19","author":"van Balen","year":"2021","journal-title":"J\u00a0Thromb Haemost"},{"key":"10.1016\/j.jtha.2023.09.029_bib18","series-title":"HealthMeasures Scoring Service","author":"Cella","year":"2007"},{"key":"10.1016\/j.jtha.2023.09.029_bib19","doi-asserted-by":"crossref","first-page":"327","DOI":"10.1179\/102453311X13085644680348","article-title":"A\u00a0novel hemostasis assay for the simultaneous measurement of coagulation and fibrinolysis","volume":"16","author":"van Geffen","year":"2011","journal-title":"Hematology"},{"key":"10.1016\/j.jtha.2023.09.029_bib20","doi-asserted-by":"crossref","first-page":"e168","DOI":"10.1016\/S2352-3026(22)00377-5","article-title":"Emicizumab in people with moderate or mild haemophilia A (HAVEN 6): a multicentre, open-label, single-arm, phase 3 study","volume":"10","author":"N\u00e9grier","year":"2023","journal-title":"Lancet Haematol"},{"key":"10.1016\/j.jtha.2023.09.029_bib21","doi-asserted-by":"crossref","first-page":"706","DOI":"10.1056\/NEJMoa2211644","article-title":"Gene therapy with etranacogene dezaparvovec for hemophilia B","volume":"388","author":"Pipe","year":"2023","journal-title":"N\u00a0Engl J Med"},{"key":"10.1016\/j.jtha.2023.09.029_bib22","doi-asserted-by":"crossref","first-page":"535","DOI":"10.1056\/NEJMoa067659","article-title":"Prophylaxis versus episodic treatment to prevent joint disease in boys with severe hemophilia","volume":"357","author":"Manco-Johnson","year":"2007","journal-title":"N\u00a0Engl J Med"},{"key":"10.1016\/j.jtha.2023.09.029_bib23","doi-asserted-by":"crossref","first-page":"192","DOI":"10.1111\/hae.14201","article-title":"Clinical phenotype of severe and moderate haemophilia: Who should receive prophylaxis and what is the target trough level?","volume":"27","author":"Collins","year":"2021","journal-title":"Haemophilia"},{"key":"10.1016\/j.jtha.2023.09.029_bib24","doi-asserted-by":"crossref","first-page":"1737","DOI":"10.1016\/j.jtha.2023.03.016","article-title":"Reclassifying hemophilia to include the definition of outcomes and phenotype as new targets","volume":"21","author":"Thachil","year":"2023","journal-title":"J\u00a0Thromb Haemost"},{"key":"10.1016\/j.jtha.2023.09.029_bib25","first-page":"12","article-title":"Mild and Moderate Hemophilia A: Neglected Conditions, Still with Unmet Needs","author":"Castaman","year":"2023","journal-title":"J\u00a0Clin Med"},{"key":"10.1016\/j.jtha.2023.09.029_bib26","doi-asserted-by":"crossref","first-page":"438","DOI":"10.1111\/j.1365-2516.2005.01124.x","article-title":"Variability in clinical phenotype of severe haemophilia: the role of the first joint bleed","volume":"11","author":"van Dijk","year":"2005","journal-title":"Haemophilia"}],"container-title":["Journal of Thrombosis and Haemostasis"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1538783623007316?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1538783623007316?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,10,27]],"date-time":"2025-10-27T03:41:48Z","timestamp":1761536508000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S1538783623007316"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,1]]},"references-count":26,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2024,1]]}},"alternative-id":["S1538783623007316"],"URL":"https:\/\/doi.org\/10.1016\/j.jtha.2023.09.029","relation":{},"ISSN":["1538-7836"],"issn-type":[{"value":"1538-7836","type":"print"}],"subject":[],"published":{"date-parts":[[2024,1]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Patients with moderate hemophilia A and B with a severe bleeding phenotype have an increased burden of disease","name":"articletitle","label":"Article Title"},{"value":"Journal of Thrombosis and Haemostasis","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.jtha.2023.09.029","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2023 International Society on Thrombosis and Haemostasis. Published by Elsevier Inc.","name":"copyright","label":"Copyright"}]}}