{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T04:21:04Z","timestamp":1791865264627,"version":"4.3.4"},"reference-count":15,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2022,12,8]],"date-time":"2022-12-08T00:00:00Z","timestamp":1670457600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100004337","name":"Roche","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004337","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Euro J of Neurology"],"published-print":{"date-parts":[[2023,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background and purpose<\/jats:title>\n                    <jats:p>The prevalence of Huntington disease (HD) has increased over time; however, there is a lack of up\u2010to\u2010date evidence documenting the economic burden of HD by disease stage. This study provides an estimate of the annual direct medical, nonmedical, and indirect costs associated with HD from participants in the Huntington's Disease Burden of Illness (HDBOI) study in five European countries and the USA.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>The HDBOI is a retrospective, cross\u2010sectional study. Data collection was conducted between September 2020 and May 2021. Participants were recruited by their HD\u2010treating physicians and categorized as early stage (ES), mid stage (MS), or advanced stage (AS) HD. Data were collected via three questionnaires: a case report form, completed by physicians who collected health care resource use associated with HD to compute direct medical cost, and optional patient and caregiver questionnaires, which included information used to compute nondirect medical and indirect costs. Country\u2010specific unit cost sources were used.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      HDBOI cost estimates were \u20ac12,663 (\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u20092094) for direct medical costs, \u20ac2984 (\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u2009359) for nondirect medical costs, and \u20ac47,576 (\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u2009436) for indirect costs. Costs are higher in patients who are at later stages of disease; for example, direct medical costs estimates were \u20ac9220 (\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u2009846), \u20ac11,885 (\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u2009701), and \u20ac18,985 (\n                      <jats:italic>n<\/jats:italic>\n                      \u00a0=\u2009547) for ES, MS, and AS, respectively. Similar trends were observed for nondirect and indirect costs. Costs show large variations between patients and countries.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Cost estimates from the HDBOI study show that people with HD and their caregivers bear a large economic burden that increases as disease progresses.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/ene.15645","type":"journal-article","created":{"date-parts":[[2022,11,24]],"date-time":"2022-11-24T13:26:59Z","timestamp":1669296419000},"page":"1109-1117","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":30,"title":["Economic burden of Huntington disease in Europe and the USA: Results from the Huntington's Disease Burden of Illness study"],"prefix":"10.1111","volume":"30","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2022-3239","authenticated-orcid":false,"given":"Idaira","family":"Rodr\u00edguez\u2010Santana","sequence":"first","affiliation":[{"name":"HCD Economics  Cheshire UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tiago","family":"Mestre","sequence":"additional","affiliation":[{"name":"Ottawa Hospital Research Institute  Ontario Ottawa Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ferdinando","family":"Squitieri","sequence":"additional","affiliation":[{"name":"Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS) Casa Sollievo della Sofferenza Research Hospital  San Giovanni Rotondo Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rosa","family":"Willock","sequence":"additional","affiliation":[{"name":"HCD Economics  Cheshire UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Astri","family":"Arnesen","sequence":"additional","affiliation":[{"name":"European Huntington Association  Kristiansand Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alison","family":"Clarke","sequence":"additional","affiliation":[{"name":"Manchester Centre for Genomic Medicine  Manchester UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Barbara","family":"D'Alessio","sequence":"additional","affiliation":[{"name":"Lega Italiana Ricerca Huntington  Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alex","family":"Fisher","sequence":"additional","affiliation":[{"name":"Birmingham and Solihull Mental Health Foundation Trust  Birmingham UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rebecca","family":"Fuller","sequence":"additional","affiliation":[{"name":"CHDI Management\/CHDI Foundation  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jamie L.","family":"Hamilton","sequence":"additional","affiliation":[{"name":"CHDI Management\/CHDI Foundation  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hayley","family":"Hubberstey","sequence":"additional","affiliation":[{"name":"Huntington's Disease Youth Organization  Kington UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cath","family":"Stanley","sequence":"additional","affiliation":[{"name":"Huntington's Disease Association  Liverpool UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Louise","family":"Vetter","sequence":"additional","affiliation":[{"name":"Huntington's Disease Society of America  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michaela","family":"Winkelmann","sequence":"additional","affiliation":[{"name":"Deutsche Huntington\u2010Hilfe  Duisburg 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