{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T04:54:58Z","timestamp":1790571298868,"version":"4.1.0"},"reference-count":40,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2025,12,8]],"date-time":"2025-12-08T00:00:00Z","timestamp":1765152000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2025,12,8]],"date-time":"2025-12-08T00:00:00Z","timestamp":1765152000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>The expansion of haemophilia treatment to include non\u2010factor prophylaxis provides new options with various benefits, risks, administration modes, and device types, necessitating trade\u2010offs in decision\u2010making.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>This study evaluated prophylactic treatment preferences of people with haemophilia and their caregivers, assessing their willingness to make trade\u2010offs among treatment attributes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A web\u2010based survey containing a discrete\u2010choice experiment (DCE) was completed by adults (\u2265 18 years old) and caregivers of children (aged 8\u201317 years) with haemophilia in the United States and United Kingdom. Participants were asked to complete 10 choice tasks, choosing their preferred prophylaxis from two hypothetical profiles defined by seven attributes. The primary analysis employed a mixed logit model, and subgroup analyses explored preference heterogeneity.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Participants were 194 adults with haemophilia (US: 150; UK: 44) and 169 caregivers (US: 150; UK: 19). Avoiding daily treatment administration had the highest attribute importance among both adults and caregivers, followed by changes in annual bleeds. Participants were willing to accept an increased risk of serious side effects or developing inhibitors, or reduced efficacy in preventing annual bleeds, to switch from intravenous to subcutaneous administration. Refrigeration requirements and the need for a second treatment for breakthrough bleeds had lower importance.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Participants highly value avoiding daily administration and reducing bleeds, and they are willing to accept increased risks or reduced efficacy to have subcutaneous rather than intravenous administration. As new treatments become available, understanding these preferences can facilitate shared decision\u2010making in treatment selection and enhance patient\u2010centred drug development and patient communication.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.70172","type":"journal-article","created":{"date-parts":[[2025,12,8]],"date-time":"2025-12-08T13:31:04Z","timestamp":1765200664000},"page":"116-130","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Patient and Caregiver Preferences for Long\u2010Term Prophylactic Treatment of Bleeds in Haemophilia: A Discrete\u2010Choice Experiment"],"prefix":"10.1111","volume":"32","author":[{"given":"Hui","family":"Lu","sequence":"first","affiliation":[{"name":"PPD Evidera Patient\u2010Centered Research Thermo Fisher Scientific  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-9416-1890","authenticated-orcid":false,"given":"Travis","family":"Gould","sequence":"additional","affiliation":[{"name":"Pfizer Inc.  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4937-0654","authenticated-orcid":false,"given":"Carmine","family":"Colavecchia","sequence":"additional","affiliation":[{"name":"Pfizer Inc.  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1795-8536","authenticated-orcid":false,"given":"Joshua","family":"Coulter","sequence":"additional","affiliation":[{"name":"Pfizer Inc.  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-2642-4827","authenticated-orcid":false,"given":"Lisa J.","family":"Wilcox","sequence":"additional","affiliation":[{"name":"Pfizer Inc.  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9586-0748","authenticated-orcid":false,"given":"Joseph C.","family":"Cappelleri","sequence":"additional","affiliation":[{"name":"Pfizer Inc.  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5792-8393","authenticated-orcid":false,"given":"Stephanie A.","family":"Christopher","sequence":"additional","affiliation":[{"name":"Pfizer Inc.  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexis","family":"Sohn","sequence":"additional","affiliation":[{"name":"Pfizer Inc.  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3129-7268","authenticated-orcid":false,"given":"Brett","family":"Hauber","sequence":"additional","affiliation":[{"name":"Pfizer Inc.  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"\u00c1lvaro A.","family":"Guti\u00e9rrez\u2010Vargas","sequence":"additional","affiliation":[{"name":"PPD Evidera Patient\u2010Centered Research Thermo Fisher Scientific  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicola","family":"Dunn","sequence":"additional","affiliation":[{"name":"Independent Consultant  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6690-8586","authenticated-orcid":false,"given":"Pratima","family":"Chowdary","sequence":"additional","affiliation":[{"name":"Katharine Dormandy Haemophilia and Thrombosis Centre Royal Free Hospital  London UK"},{"name":"Department of Haematology University College  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5886-1933","authenticated-orcid":false,"given":"Jennifer A.","family":"Whitty","sequence":"additional","affiliation":[{"name":"PPD Evidera Patient\u2010Centered Research Thermo Fisher Scientific  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,12,8]]},"reference":[{"key":"e_1_2_12_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(24)02139-1"},{"key":"e_1_2_12_3_1","doi-asserted-by":"publisher","DOI":"10.2147\/JBM.S133616"},{"key":"e_1_2_12_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14046"},{"key":"e_1_2_12_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/rth2.12695"},{"key":"e_1_2_12_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13221"},{"key":"e_1_2_12_7_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S139851"},{"key":"e_1_2_12_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2012.02770.x"},{"key":"e_1_2_12_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.18495"},{"key":"e_1_2_12_10_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1803550"},{"key":"e_1_2_12_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1538-7836.2011.04611.x"},{"key":"e_1_2_12_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/S2352-3026(24)00307-7"},{"key":"e_1_2_12_13_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2023021864"},{"issue":"47","key":"e_1_2_12_14_1","first-page":"791","article-title":"Treatment Options in Hemophilia","volume":"116","author":"Miesbach W.","year":"2019","journal-title":"Deutsches Arzteblatt International"},{"key":"e_1_2_12_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14037"},{"key":"e_1_2_12_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14928"},{"key":"e_1_2_12_17_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0256521"},{"key":"e_1_2_12_18_1","doi-asserted-by":"publisher","DOI":"10.2165\/00019053-200826080-00004"},{"key":"e_1_2_12_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2016.04.004"},{"key":"e_1_2_12_20_1","doi-asserted-by":"publisher","DOI":"10.1186\/s41687-023-00550-6"},{"key":"e_1_2_12_21_1","doi-asserted-by":"publisher","DOI":"10.1136\/jamia.1998.0050257"},{"issue":"9","key":"e_1_2_12_22_1","first-page":"682","article-title":"Shared Decision Making in Preventive Health Care: What It Is; What It Is Not","volume":"63","author":"Grad R.","year":"2017","journal-title":"Canadian Family Physician"},{"key":"e_1_2_12_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40273-018-0734-2"},{"key":"e_1_2_12_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2010.11.013"},{"key":"e_1_2_12_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40271-024-00672-z"},{"key":"e_1_2_12_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/hec.1369"},{"key":"e_1_2_12_27_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40271-020-00475-y"},{"key":"e_1_2_12_28_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1755-5345(13)70051-4"},{"key":"e_1_2_12_29_1","doi-asserted-by":"publisher","DOI":"10.1080\/14737167.2017.1389648"},{"key":"e_1_2_12_30_1","doi-asserted-by":"publisher","DOI":"10.1162\/003465398557735"},{"key":"e_1_2_12_31_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0191-2615(02)00046-2"},{"key":"e_1_2_12_32_1","doi-asserted-by":"publisher","DOI":"10.1177\/193229681100500633"},{"key":"e_1_2_12_33_1","unstructured":"M.Benton L.Shi A. M.Patel et\u00a0al. \u201cPB0245 Evaluating Patient and Caregiver Treatment Preferences in Hemophilia Using a Using a Discrete Choice Experiment \u201dXXVII Congress of the International Society on Thrombosis and Haemostasis.Melbourne Australia 270\u2013271."},{"key":"e_1_2_12_34_1","doi-asserted-by":"publisher","DOI":"10.2147\/PPA.S24752"},{"key":"e_1_2_12_35_1","doi-asserted-by":"publisher","DOI":"10.33393\/dti.2021.2342"},{"key":"e_1_2_12_36_1","doi-asserted-by":"publisher","DOI":"10.1185\/03007995.2011.598500"},{"key":"e_1_2_12_37_1","doi-asserted-by":"publisher","DOI":"10.3810\/pgm.2013.05.2656"},{"key":"e_1_2_12_38_1","first-page":"1687","article-title":"Patient and Parent Preferences for Characteristics of Prophylactic Treatment in Hemophilia","volume":"9","author":"Furlan R.","year":"2015","journal-title":"Patient Preference and Adherence"},{"key":"e_1_2_12_39_1","doi-asserted-by":"publisher","DOI":"10.1142\/9789814417358_0006"},{"key":"e_1_2_12_40_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2018.07.876"},{"key":"e_1_2_12_41_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2019.04.1924"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70172","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.70172","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70172","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T03:55:39Z","timestamp":1790567739000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.70172"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,12,8]]},"references-count":40,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,1]]}},"alternative-id":["10.1111\/hae.70172"],"URL":"https:\/\/doi.org\/10.1111\/hae.70172","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,12,8]]},"assertion":[{"value":"2025-07-22","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-11-06","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-12-08","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}