{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T15:32:33Z","timestamp":1756308753216,"version":"3.40.5"},"reference-count":30,"publisher":"Public Library of Science (PLoS)","issue":"12","license":[{"start":{"date-parts":[[2022,12,30]],"date-time":"2022-12-30T00:00:00Z","timestamp":1672358400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:sec id=\"sec001\">\n<jats:title>Objectives<\/jats:title>\n<jats:p>To describe education level and employment status among adults with Loeys-Dietz syndrome and vascular Ehlers-Danlos syndrome, and explore factors related to work participation.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec002\">\n<jats:title>Materials and methods<\/jats:title>\n<jats:p>Cross-sectional postal survey in 2018. Individuals with molecularly verified diagnosis were recruited through a National Resource Centre for Rare Disorders. A study specific questionnaire included topics on disease burden and validated instruments regarding education level, employment, pain, fatigue, psychological distress, and satisfaction with life.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec003\">\n<jats:title>Results<\/jats:title>\n<jats:p>Fifty persons (56% women) aged 18\u201367 years, participated. Almost 60% reported education level \u226413 years. Two thirds (66%) received disability benefits, 21 (42%) had full-time disability pension. The median age at ending work was 41 years. Full-time employed and students were younger (p = 0.014), less fatigued (p = 0.035), had less sleep problems (p = 0.028) and higher satisfaction with life (p&lt;0.001) than those who received disability pension. A third (32%) were currently or used to be in sedentary work, and 68% currently had or used to be in practical work requiring much standing and walking (23%), much walking and lifting (34%) or heavy manual work (11%).<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec004\">\n<jats:title>Conclusions<\/jats:title>\n<jats:p>There is a potential that more adults with these diagnoses can sustain employment for more years. Health and social service follow-up routines and future studies should include details on employment perspectives to reveal those at risk of poor employment and to identify modifiable factors for work participation.<\/jats:p>\n<\/jats:sec>","DOI":"10.1371\/journal.pone.0279848","type":"journal-article","created":{"date-parts":[[2022,12,30]],"date-time":"2022-12-30T18:57:17Z","timestamp":1672426637000},"page":"e0279848","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":3,"title":["Education and employment status among adults with Loeys-Dietz syndrome and vascular Ehlers-Danlos syndrome in Norway, a questionnaire based study"],"prefix":"10.1371","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1396-6715","authenticated-orcid":true,"given":"Heidi","family":"Johansen","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gry","family":"Velvin","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ingeborg B.","family":"Lidal","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2022,12,30]]},"reference":[{"key":"pone.0279848.ref001","doi-asserted-by":"crossref","first-page":"158","DOI":"10.1016\/j.ijcard.2022.05.065","article-title":"Clinical features and complications of Loeys-Dietz syndrome: A systematic review","volume":"362","author":"P Gouda","year":"2022","journal-title":"Int J Cardiol"},{"issue":"1","key":"pone.0279848.ref002","doi-asserted-by":"crossref","first-page":"40","DOI":"10.1002\/ajmg.c.31553","article-title":"Diagnosis, natural history, and management in vascular Ehlers-Danlos syndrome","volume":"175","author":"PH Byers","year":"2017","journal-title":"Am J Med Genet C"},{"issue":"6","key":"pone.0279848.ref003","doi-asserted-by":"crossref","first-page":"582","DOI":"10.21037\/acs.2017.11.03","article-title":"Differences in manifestations of Marfan syndrome, Ehlers-Danlos syndrome, and Loeys-Dietz syndrome","volume":"6","author":"JAN Meester","year":"2017","journal-title":"Ann Cardiothorac Surg"},{"issue":"1","key":"pone.0279848.ref004","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1002\/ajmg.a.61396","article-title":"Adults with Loeys-Dietz syndrome and vascular Ehlers-Danlos syndrome: A cross-sectional study of health burden perspectives","volume":"182","author":"H Johansen","year":"2020","journal-title":"Am J Med Genet A"},{"volume-title":"GeneReviews((R))","year":"2017","author":"DM Milewicz","key":"pone.0279848.ref005"},{"issue":"1","key":"pone.0279848.ref006","first-page":"34","article-title":"Quantifying health status and function in Marfan syndrome","volume":"25","author":"SS Rao","year":"2016","journal-title":"J Surg Orthopaedic Adv"},{"issue":"12","key":"pone.0279848.ref007","doi-asserted-by":"crossref","first-page":"3082","DOI":"10.1002\/ajmg.a.37370","article-title":"Work participation in adults with Marfan syndrome: Demographic characteristics, MFS related health symptoms, chronic pain, and fatigue","volume":"167","author":"G Velvin","year":"2015","journal-title":"Am J Med Genet A"},{"issue":"23","key":"pone.0279848.ref008","doi-asserted-by":"crossref","first-page":"2821","DOI":"10.1016\/j.jacc.2017.04.026","article-title":"Marfan syndrome and quality of life in the GenTAC registry","volume":"69","author":"JZ Goldfinger","year":"2017","journal-title":"J Am Coll Cardiol"},{"key":"pone.0279848.ref009","first-page":"1","article-title":"Male\u2013female differences in quality of life and coping style in patients with Marfan syndrome and hereditary thoracic aortic diseases","volume":"00","author":"CG Thijssen","year":"2020","journal-title":"J Genet Couns"},{"key":"pone.0279848.ref010","unstructured":"NAV Information about NAV`s services and benefits- nav.no.[Internet] [cited 2021 Dec 15] https:\/\/www.nav.no\/en\/home\/benefits-and-services\/information-about-nav-s-services-and-benefits"},{"key":"pone.0279848.ref011","unstructured":"K\u00f8ber T, B\u00f8 TP. 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