{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,4]],"date-time":"2026-06-04T09:49:55Z","timestamp":1780566595287,"version":"3.54.1"},"reference-count":31,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2016,5,13]],"date-time":"2016-05-13T00:00:00Z","timestamp":1463097600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"BURQOL-RD project","award":["A101205"],"award-info":[{"award-number":["A101205"]}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["J intellect Disabil Res"],"published-print":{"date-parts":[[2016,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>To date, there has been no published comprehensive estimation of costs related to Prader\u2013Willi syndrome (PWS). Our objective was therefore to provide data on the economic burden and health\u2010related quality of life associated with PWS in France in order to raise awareness of the repercussions on individuals suffering from this syndrome and on caregivers as well as on the health and social care systems.<\/jats:p><\/jats:sec><jats:sec><jats:title>Method<\/jats:title><jats:p>A retrospective cross\u2010sectional study was carried out on 51 individuals recruited through the French PWS patient association. Data on their demographic characteristics and resource use were obtained from an online questionnaire, and costs were estimated by a bottom\u2010up approach. The EQ\u20105D\u20105L health questionnaire was used to measure the health\u2010related quality of life of individuals suffering from PWS and their caregivers.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The average annual cost of PWS was estimated at \u20ac58\u2009890 per individual, with direct healthcare accounting for \u20ac42\u2009299, direct non\u2010healthcare formal costs \u20ac13\u2009865 and direct non\u2010healthcare informal costs \u20ac8459. The main contributors to PWS costs were hospitalisations and social services. Indirect costs resulting from loss of productivity in the labour market was \u20ac32\u2009542 for adults suffering from PWS. Mean EQ\u20105D utility scores were 0.4 for individuals with PWS and 0.7 for caregivers.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Prader\u2013Willi syndrome represents a major economic burden from a societal perspective and has a significant impact on health\u2010related quality of life both for individuals suffering from PWS and for their caregivers in France. These results underscore the need to develop tailored policies targeted at improving care. Likewise, a larger study collecting a broader range of medical characteristics should be undertaken to achieve more precise estimations.<\/jats:p><\/jats:sec>","DOI":"10.1111\/jir.12288","type":"journal-article","created":{"date-parts":[[2016,5,13]],"date-time":"2016-05-13T05:03:32Z","timestamp":1463115812000},"page":"879-890","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":22,"title":["Economic burden and health\u2010related quality of life associated with Prader\u2013Willi syndrome in France"],"prefix":"10.1111","volume":"60","author":[{"given":"K.","family":"Chevreul","sequence":"first","affiliation":[{"name":"Sorbonne Paris Cit\u00e9 Univ Paris Diderot  Paris France"},{"name":"Inserm ECEVE  Paris France"},{"name":"URC\u2010Eco AP\u2010HP  Paris France"},{"name":"Univ Paris\u2010Est Cr\u00e9teil  Cr\u00e9teil France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K.","family":"Berg Brigham","sequence":"additional","affiliation":[{"name":"URC\u2010Eco AP\u2010HP  Paris France"},{"name":"Univ Paris\u2010Est Cr\u00e9teil  Cr\u00e9teil France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.\u2010C.","family":"Cl\u00e9ment","sequence":"additional","affiliation":[{"name":"URC\u2010Eco AP\u2010HP  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C.","family":"Poitou","sequence":"additional","affiliation":[{"name":"Institute of Cardiometabolism and Nutrition, ICAN, AP\u2010HP, Nutrition Department and French Reference Centre for Prader\u2010Willi Syndrome University Pierre et Marie Curie\u2010Paris 6  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.","family":"Tauber","sequence":"additional","affiliation":[{"name":"Reference Centre for Prader\u2010Willi Syndrome, Children Hospital, Department of Endocrinology, Obesity, Bone Diseases, Genetics and Gynecology Toulouse Hospital  Toulouse France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"Members of the BURQOL\u2010RD Research Network listed in the Online Appendix","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2016,5,13]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.healthpol.2014.12.016"},{"key":"e_1_2_9_3_1","unstructured":"Assurance maladie(2012a)Base des m\u00e9dicaments et informations tarifaires."},{"key":"e_1_2_9_4_1","unstructured":"Assurance maladie(2012b)Classification commune des actes m\u00e9dicaux."},{"key":"e_1_2_9_5_1","unstructured":"Assurance maladie(2012c)Liste des produits et prestations."},{"key":"e_1_2_9_6_1","unstructured":"Assurance maladie(2012d)Table nationale de codage de biologie."},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2788.2006.00878.x"},{"key":"e_1_2_9_8_1","volume-title":"Medical Research What's It Worth? 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