{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T23:33:02Z","timestamp":1787787182993,"version":"build-2784847793"},"reference-count":18,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2022,6,21]],"date-time":"2022-06-21T00:00:00Z","timestamp":1655769600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2022,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>GOAL\u2010H\u0113m is a novel, haemophilia\u2010specific, patient\u2010centred outcome measure (PCOM) based on goal attainment scaling, allowing people with haemophilia (PwH) to set and monitor the attainment of individualized goals for treatment.<\/jats:p><\/jats:sec><jats:sec><jats:title>Aim<\/jats:title><jats:p>To provide a thorough overview of the creation, validation, and development of GOAL\u2010H\u0113m.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Clinician workshops were held to develop a haemophilia\u2010specific goal menu. Qualitative data from semistructured interviews with PwH and their caregivers guided further revisions to the goal menu (i.e., goal domains and descriptors). A feasibility study was performed including a 12\u2010week, prospective, noninterventional evaluation involving clinicians and PwH at four US haemophilia treatment centres. Finally, the Patient Voice Study gathered feedback from PwH and their caregivers via an online survey, interviews, and a focus group.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The feasibility study validated GOAL\u2010H\u0113m with successful outcomes in construct\/content validity and responsiveness, including a large effect in patient\u2010 and clinician\u2010rated goal attainments. The Patient Voice Study led to significant refinement of GOAL\u2010H\u0113m goals and descriptors, resulting in a more straightforward and relatable menu for PwH and their caregivers. Overall, GOAL\u2010H\u0113m captured qualitative data in areas important to PwH and employed quantitative methods to evaluate meaningful changes in those areas. The individualized tool was well equipped to handle the complex and chronic nature of haemophilia and was endorsed by PwH, their caregivers, and clinicians.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The GOAL\u2010H\u0113m development journey may serve as a roadmap for other PCOMs in a variety of settings, including clinical studies, haemophilia treatment centres for care planning, and as a tool to gather real\u2010world evidence.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.14608","type":"journal-article","created":{"date-parts":[[2022,6,21]],"date-time":"2022-06-21T20:55:05Z","timestamp":1655844905000},"page":"784-795","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":16,"title":["The GOAL\u2010H\u0113m journey: Shared decision making and patient\u2010centred outcomes"],"prefix":"10.1111","volume":"28","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9415-9543","authenticated-orcid":false,"given":"Jonathan C.","family":"Roberts","sequence":"first","affiliation":[{"name":"Bleeding &amp; Clotting Disorders Institute  Peoria Illinois USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sharon","family":"Richardson","sequence":"additional","affiliation":[{"name":"Astellas Pharma Global Development (APGD)  Northbrook Illinois USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Moses E.","family":"Miles","sequence":"additional","affiliation":[{"name":"American Thrombosis and Hemostasis Network  Rochester New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Justin","family":"Stanley","sequence":"additional","affiliation":[{"name":"Ardea Outcomes  Halifax Nova Scotia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chere A. T.","family":"Chapman","sequence":"additional","affiliation":[{"name":"Ardea Outcomes  Halifax Nova Scotia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Denne","sequence":"additional","affiliation":[{"name":"Takeda  Cambridge Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jorge","family":"Caicedo","sequence":"additional","affiliation":[{"name":"Takeda  Cambridge Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenneth","family":"Rockwood","sequence":"additional","affiliation":[{"name":"Ardea Outcomes  Halifax Nova Scotia Canada"},{"name":"Dalhousie University  Halifax Nova Scotia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2805-1016","authenticated-orcid":false,"given":"Michael","family":"Recht","sequence":"additional","affiliation":[{"name":"American Thrombosis and Hemostasis Network  Rochester New York USA"},{"name":"The Hemophilia Center at Oregon Health &amp; Science University  Portland Oregon USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,6,21]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13066"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1182\/bloodadvances.2018020552"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejh.12323"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13454"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF01530764"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1093\/geront\/38.6.735"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0885-3924(98)00106-7"},{"key":"e_1_2_11_9_1","unstructured":"NeufeldEJ MunnJ HonigD DenneM RichardsonS RockwoodK Development of a novel patient\u2010centered outcome measure in hemophilia using goal attainment scaling. Presented at: National Hemophilia Foundation (NHF) 67th Annual Meeting;Grapevine TX USA.2015."},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/rth2.12655"},{"issue":"4","key":"e_1_2_11_11_1","first-page":"3","article-title":"Goal Attainment Scaling for life \u2010 Hemophilia (GOAL\u2010Hem): an innovative patient\u2010reported outcome measure","volume":"22","author":"Recht M","year":"2016","journal-title":"Haemophilia"},{"key":"e_1_2_11_12_1","unstructured":"LattimoreS SchwartzE GoldstineJ FayS ChapmanC Patient and clinician experience of using goal attainment scaling for hemophilia (GAS\u2010Hem) an innovative patient\u2010centered outcome measure. Presented at: National Hemophilia Foundation (NHF) Annual Meeting;Chicago IL USA.2017."},{"key":"e_1_2_11_13_1","unstructured":"StanleyJ DunnT CaicedoJ DenneM RobertsJ RockwoodK Implementing GOAL\u2010H\u0113m a goal attainment scaling instrument. Presented at: Thrombosis & Hemostasis Summit of North America Virtual Meeting.2020."},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1186\/s41687-019-0110-9"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1159\/000054761"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1186\/s41687-020-00196-8"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1159\/000178757"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1186\/s41687-019-0100-y"},{"key":"e_1_2_11_19_1","unstructured":"Clinicaltrials.gov. ATHN Transcends: a natural history study of non\u2010neoplastic hematologic disorders (NCT04398628). Available at:https:\/\/clinicaltrials.gov\/ct2\/show\/NCT04398628?term=NCT04398628&draw=2&rank=1. Accessed July 23 2021."}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.14608","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.14608","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.14608","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,22]],"date-time":"2023-08-22T08:43:08Z","timestamp":1692693788000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.14608"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,6,21]]},"references-count":18,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2022,9]]}},"alternative-id":["10.1111\/hae.14608"],"URL":"https:\/\/doi.org\/10.1111\/hae.14608","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,6,21]]},"assertion":[{"value":"2022-02-14","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-06-02","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-06-21","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}