{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,15]],"date-time":"2026-09-15T01:58:54Z","timestamp":1789437534364,"version":"build-2803163510"},"reference-count":48,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2015,9,14]],"date-time":"2015-09-14T00:00:00Z","timestamp":1442188800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2016,1]]},"abstract":"<jats:sec><jats:label\/><jats:p>Clinical interventions and research have mostly focused on the orthopedic, genetic, and pharmacological outcomes of individuals with osteogenesis imperfecta (OI), and although quality of life (QoL) has gained recognition as an important patient\u2010outcome, it has received little attention in individuals with OI. This mixed\u2010methods systematic review of the literature included five search engines and identified a total of 212 articles. Once study eligibility was reviewed, 10 studies met the inclusion criteria and were included in this mixed\u2010methods review (9 quantitative and 1 qualitative). Among the 10 included QoL studies, six reported on children with OI, three on adults with OI, and one on the parents of children with OI. Physical QoL in children and adults with OI appears to be less than that of the general population, with individuals with more severe OI types reporting worse QoL. On the other hand, mental and psychosocial QoL is the same or better in individuals with OI than that of the general population. Pain, scoliosis activity limitations and participation restrictions due to decreased function are associated with lower levels of physical QoL. Researchers must agree on a definition of QoL as it relates to OI and use validated measures appropriate for evaluating QoL in OI. Pediatric studies should consider both the child and the parent's QOL perceptions as these may differ. QoL in the adult population should not be dismissed in order to offer proper client\u2010centered interventions throughout the lifespan. \u00a9 2015 Wiley Periodicals, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/ajmg.a.37377","type":"journal-article","created":{"date-parts":[[2015,9,14]],"date-time":"2015-09-14T06:08:13Z","timestamp":1442210893000},"page":"62-76","source":"Crossref","is-referenced-by-count":74,"title":["Quality of life in osteogenesis imperfecta: A mixed\u2010methods systematic review"],"prefix":"10.1002","volume":"170","author":[{"given":"N.","family":"Dahan\u2010Oliel","sequence":"first","affiliation":[{"name":"Shriners Hospital for Children Canada"},{"name":"School of Physical and Occupational Therapy Faculty of Medicine McGill University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Oliel","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children Canada"},{"name":"School of Physical and Occupational Therapy Faculty of Medicine McGill University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Tsimicalis","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children Canada"},{"name":"Ingram School of Nursing Faculty of Medicine McGill University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K.","family":"Montpetit","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"F.","family":"Rauch","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children Canada"},{"name":"Department of Pediatrics Faculty of Medicine McGill University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M. 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