{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,30]],"date-time":"2026-10-30T07:50:05Z","timestamp":1793346605694,"version":"4.5.0"},"reference-count":20,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2024,9,5]],"date-time":"2024-09-05T00:00:00Z","timestamp":1725494400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2024,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>Currently, no quality\u2010of\u2010life instrument exists that captures the full experience of the mental health outlook (MHO), a coreHEM core outcome, in people with haemophilia, including the potential transformational experience of receiving gene therapy.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>To describe the methods used to develop a content validated patient\u2010reported outcome measure (PROM) that measures MHO for people with haemophilia.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A conceptual framework, developed from a literature\/evidence review, was used to create an interview guide and draft a questionnaire. Males aged 15 or older with severe\/moderate haemophilia were eligible to participate in concept elicitation or cognitive debriefing interviews. The conceptual framework was refined based on a thematic analysis of concept elicitation interviews and PROM items were developed from the conceptual framework. Cognitive debriefing sessions that prioritised relevance and understanding of the PROM were held in two rounds; items were updated iteratively.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>A conceptual framework with five domains (stigma, anxiety, depression, life interference and identity) was constructed from over 300 identified MHO concepts. Fifty\u2010three participants took part in interviews. After 32 concept elicitation interviews, the framework was updated by including eight new sub\u2010concepts and eliminating two. Updates to the questionnaire included items added or removed and improved wording. The final coreHEM MHO PROM has 26 questions in two sections (general mental health associated with haemophilia, and a gene therapy section).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The instrument is content\u2010validated and can be used as an exploratory outcome. MHO scores can be measured and compared to give more insight into patient quality of life.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.15085","type":"journal-article","created":{"date-parts":[[2024,9,6]],"date-time":"2024-09-06T00:28:44Z","timestamp":1725582524000},"page":"1309-1320","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Development of the coreHEM mental health patient\u2010reported outcome measure \u2013 A novel mental health outlook measure for people with haemophilia"],"prefix":"10.1111","volume":"30","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1789-6635","authenticated-orcid":false,"given":"Elizabeth","family":"Clearfield","sequence":"first","affiliation":[{"name":"Center for Medical Technology Policy  Baltimore USA"},{"name":"Institute for Policy Advancement Ltd  Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hsing\u2010Yuan","family":"Chang","sequence":"additional","affiliation":[{"name":"Center for Medical Technology Policy  Baltimore USA"},{"name":"Department of Pediatrics Chang Gung Memorial Hospital  Taoyuan Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ellen M.","family":"Janssen","sequence":"additional","affiliation":[{"name":"Center for Medical Technology Policy  Baltimore USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tabassum","family":"Majid","sequence":"additional","affiliation":[{"name":"Erickson School of Aging Studies University of Maryland  Baltimore USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donna A.","family":"Messner","sequence":"additional","affiliation":[{"name":"Center for Medical Technology Policy  Baltimore USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8372-4474","authenticated-orcid":false,"given":"Donna","family":"Coffin","sequence":"additional","affiliation":[{"name":"World Federation of Hemophilia  Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohit","family":"Jain","sequence":"additional","affiliation":[{"name":"BioMarin Pharmaceuticals Ltd  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul E.","family":"Monahan","sequence":"additional","affiliation":[{"name":"CSL Behring  King of Prussia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7927-6418","authenticated-orcid":false,"given":"Leonard A.","family":"Valentino","sequence":"additional","affiliation":[{"name":"National Bleeding Disorders Foundation  New York USA"},{"name":"Hemophilia and Thrombophilia Center, Rush Medical College Rush University Chicago USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michelle","family":"Witkop","sequence":"additional","affiliation":[{"name":"National Bleeding Disorders Foundation  New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0934-0680","authenticated-orcid":false,"given":"Mark W.","family":"Skinner","sequence":"additional","affiliation":[{"name":"Institute for Policy Advancement Ltd  Washington USA"},{"name":"Department of Health Resource Methods Evidence, and Impact McMaster University  Hamilton Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2024,9,5]]},"reference":[{"key":"e_1_2_11_2_1","volume-title":"GeneReviews\u00ae [Internet]","author":"Konkle BA"},{"key":"e_1_2_11_3_1","volume-title":"GeneReviews\u00ae [Internet]","author":"Konkle BA"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0042-1756188"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1055\/a-1175-6530"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ymthe.2023.03.001"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14755"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12995"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.5581\/1516-8484.20130108"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14413"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1080\/17474086.2023.2171981"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1089\/hum.2023.112"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13504"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13456"},{"key":"e_1_2_11_15_1","first-page":"56","article-title":"International haemophilia access strategy council. Haemophilia gene therapy (GT) outcomes: integration of the haemophilia value framework with coreHEM","volume":"21","author":"Skinner M","year":"2022","journal-title":"Haemophilia"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2011.06.014"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jval.2011.06.013"},{"key":"e_1_2_11_18_1","volume-title":"Guidance for Industry: Patient\u2010Reported Outcome Measures: Use in Medical Product Development to Support Labeling Claims","author":"U.S. Department of Health and Human Services Food and Drug Administration (FDA), Center for Drug Evaluation and Research (CDER), Center for Biologics Evaluation and Research (CBER), Center for Devices and Radiological Health (CDRH)","year":"2009"},{"issue":"4","key":"e_1_2_11_19_1","first-page":"62","article-title":"Developing a patient reported outcome measure to explore mental health outlook in people with hemophilia: development of a conceptual framework","volume":"26","author":"Clearfield E","year":"2020","journal-title":"Haemophilia"},{"key":"e_1_2_11_20_1","unstructured":"ClearfieldE JanssenEM ReedS ChangH\u2010Y WitkopM SkinnerMW.A review of current patient reported outcome measures used to assess mental health in people with hemophilia. Poster presented at the National Bleeding Disorders Foundation (NBDF) Annual Meeting. (August 30\u2010September 4 2020)."},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.7326\/M19-1208"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.15085","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,12,19]],"date-time":"2024-12-19T23:58:59Z","timestamp":1734652739000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.15085"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,9,5]]},"references-count":20,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2024,11]]}},"alternative-id":["10.1111\/hae.15085"],"URL":"https:\/\/doi.org\/10.1111\/hae.15085","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,9,5]]},"assertion":[{"value":"2024-06-17","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-07-29","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-09-05","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}