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Research methodology, demographics and initial insights from the survey have been previously reported. The cost of illness (healthcare resource use, productivity loss, out-of-pocket spending) and drivers of the economic impact of OI are reported here.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Methods<\/jats:title>\n                <jats:p>IMPACT was an international mixed-methods online survey in eight languages (fielded July\u2013September 2021) targeting adults (aged\u2009\u2265\u200918 years) or adolescents (aged\u2009\u2265\u200912\u201317 years) with OI, caregivers with or without OI and other close relatives. Survey domains included demographics, socioeconomic factors, clinical characteristics, treatment patterns, quality of life and health economics. The health economic domain for adults, which included questions on healthcare resource use, productivity loss and out-of-pocket spending, was summarised. Regression and pairwise analyses were conducted to identify independent drivers and associations with respondent characteristics.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Results<\/jats:title>\n                <jats:p>Overall, 1,440 adults with OI responded to the survey. Respondents were mostly female (70%)\u00a0and from Europe (63%) with a median age of 43 years. Within a 12-month period, adults with OI reported visiting a wide range of healthcare professionals. Two-thirds (66%) of adults visited a hospital, and one-third (33%) visited\u00a0the emergency department. The mean total number of diagnostic tests undergone by adults within these 12 months was 8.0. Adults had undergone a mean total of 11.8 surgeries up to the time point of the survey. The proportions of adults using queried consumables or services over 12 months ranged from 18\u201382%, depending on the type of consumable or service. Most adults (58%) were in paid employment, of which nearly one-third (29%) reported missing a workday. Of the queried expenses, the mean total out-of-pocket spending in 4\u00a0weeks was \u20ac191. Respondent characteristics such as female sex, more severe self-reported OI and the experience of fractures were often associated with increased economic burden.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>IMPACT provides novel insights into the substantial cost of illness associated with OI on individuals, healthcare systems and society at large. Future analyses will provide insights into country-specific economic impact, humanistic impact and the healthcare journey of individuals with OI.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s13023-024-03218-6","type":"journal-article","created":{"date-parts":[[2024,6,3]],"date-time":"2024-06-03T12:02:01Z","timestamp":1717416121000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["The IMPACT Survey: the economic impact of osteogenesis imperfecta in adults"],"prefix":"10.1186","volume":"19","author":[{"given":"Tracy","family":"Hart","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ingunn","family":"Westerheim","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Taco","family":"van Welzenis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Oliver","family":"Semler","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cathleen","family":"Raggio","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank","family":"Rauch","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-3366-3808","authenticated-orcid":false,"given":"Ruby","family":"Dadzie","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Samantha","family":"Prince","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lena Lande","family":"Wekre","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,6,3]]},"reference":[{"key":"3218_CR1","first-page":"w14322","volume":"146","author":"A Bregou Bourgeois","year":"2016","unstructured":"Bregou Bourgeois A, et al. 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Wickenstones. CR has received payments for their contributions to the present manuscript. TH serves as the Chief Executive Officer of the Osteogenesis Imperfecta Foundation and has received unrestricted educational grants from Mereo BioPharma Group London, United Kingdom and Ultragenyx Pharmaceuticals Inc, Novato, USA. OS has participated in a national advisory board for Mereo BioPharma Group London, United Kingdom and has received study contracts for clinical studies from Ultragenyx Pharmaceuticals Inc, Novato, USA. FR has received study contracts for experimental preclinical studies with Precithera Inc, Quebec, Canada, Mesentech Inc, Vancouver, Canada and Catabasis Pharmaceuticals Inc, Cambridge, USA. He has participated in advisory boards for Ultragenyx Pharmaceuticals Inc, Novato, USA, Sanofi S.A. Paris, France, Novartis International AG, Basel, Switzerland and Mereo BioPharma Group, London, United Kingdom. FR has received a speaker fee from Ultragenyx Pharmaceuticals Inc, Novato, USA for a lecture and received a donation of experimental drugs for a preclinical study from Acceleron Pharma Inc, Cambridge, USA. CR received an institutional grant from and has been a speaker for BioMarin Pharmaceuticals Inc, Novato, California, has participated in advisory boards for Ultragenyx Pharmaceuticals Inc, Novato, USA. She has received an institutional grant from and is a consultant for Nextcure, Beltville, MD, USA. CR sits on the board of directors of the Osteogenesis Imperfecta Foundation. CR has also participated in an advisory board for Mereo BioPharma Group, London, United Kingdom. TW and IW hold leadership positions in the Osteogenesis Imperfecta Federation Europe, which has received grants from Mereo BioPharma Group, London, United Kingdom (unrestricted), Ultragenyx Pharmaceuticals Inc, Novato, USA (restricted grant for a conference), Quince Therapeutics Inc, San Francisco, CA, USA (donation for a conference), UCB (restricted grant to conference), Angitia Biopharmaceuticals Inc., Woodland Hills, CA, USA (donation to conference), Azafaros BV, Naarden, the Netherlands (donation to conference), Alexion Inc. Boston, MA, USA, (sponsorship of conference); Pega Medical Inc, Laval, Quebec, Canada (donation), Sanofi S.A. Paris, France (payment to IW for engagement in steering committee; donated to OIFE). IW has also received payment as a panellist from Takeda, Tokyo, Japan.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"222"}}