The psychosocial experience of individuals living with osteogenesis imperfecta: a mixed-methods systematic review

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dc.citation.issue8
dc.citation.volume25
dc.contributor.authorTsimicalis, Argerie
dc.contributor.authorDenis-Larocque, Gabrielle
dc.contributor.authorMichalovic, Alisha
dc.contributor.authorLepage, Carolann
dc.contributor.authorWilliams, Karl
dc.contributor.authorYao, Tian-Ran
dc.contributor.authorPalomo, Telma [UNIFESP]
dc.contributor.authorDahan-Oliel, Noemi
dc.contributor.authorLe May, Sylvie
dc.contributor.authorRauch, Frank
dc.coverageDordrecht
dc.date.accessioned2020-08-14T13:44:03Z
dc.date.available2020-08-14T13:44:03Z
dc.date.issued2016
dc.description.abstractOsteogenesis imperfecta (OI) is a genetic disorder (prevalence: 1:10,000), leading to bone fragility, frequent fractures, and varying degrees of physical limitations. Despite a substantial amount of research on the genetics, pathophysiology, and treatments related to OI, there remains a paucity of knowledge concerning the lived psychosocial experience of the OI population. This mixed-methods systematic review aimed to review, appraise, and synthesize the literature on the psychosocial experience of children and adults with OI with the goal of identifying implications for research, practice, and policy-making. Using a systematic methodology, quantitative, qualitative, and mixed-methods studies were accessed through database searching, screened, assessed for eligibility, and appraised. Data from the selected studies fulfilling the eligibility and quality criteria were extracted and synthesized using thematic analysis with an inductive approach. A total of four qualitative and 20 quantitative studies, with various study designs and methodologies ranging in quality, were included in the review (n = 800en
dc.description.abstractcomprising 610 children and 175 adults with OI types I, III, IV, and V, ten parents and five healthcare professionals). Six themes were identified: intellectual feats, isolation and feeling different, fear of fractures, coping with challenges, adapting by learning new skills, and social relationships. These findings highlighted key aspects of the experiences of children and adults with OI and will be essential for improving the quality and direction of research, tailoring clinical interventions addressing the psychosocial needs and quality of life of individuals with OI, and raising awareness among caregivers, healthcare professionals, administrators, and policy-makers associated with the OI population.en
dc.description.affiliationMcGill Univ, Ingram Sch Nursing, Fac Med, Montreal, PQ H3A 2A7, Canada
dc.description.affiliationShriners Hosp Children Canada, Montreal, PQ, Canada
dc.description.affiliationUniv Fed Sao Paulo, Div Endocrinol, Bone & Mineral Unit, So Paulo, Brazil
dc.description.affiliationMcGill Univ, Sch Phys & Occupat Therapy, Fac Med, Montreal, PQ, Canada
dc.description.affiliationCHU Ste Justine, Montreal, PQ, Canada
dc.description.affiliationUniv Montreal, Fac Nursing, Montreal, PQ, Canada
dc.description.affiliationUnifespUniv Fed Sao Paulo, Div Endocrinol, Bone & Mineral Unit, So Paulo, Brazil
dc.description.sourceWeb of Science
dc.description.sponsorshipNewton Foundation
dc.description.sponsorshipTunis Shriners
dc.description.sponsorshipQuebec Nursing Intervention Research Network
dc.format.extent1877-1896
dc.identifierhttp://dx.doi.org/10.1007/s11136-016-1247-0
dc.identifier.citationQuality Of Life Research. Dordrecht, v. 25, n. 8, p. 1877-1896, 2016.
dc.identifier.doi10.1007/s11136-016-1247-0
dc.identifier.issn0962-9343
dc.identifier.urihttps://repositorio.unifesp.br/handle/11600/57498
dc.identifier.wosWOS:000380005900002
dc.language.isoeng
dc.publisherSpringer
dc.relation.ispartofQuality Of Life Research
dc.rightsinfo:eu-repo/semantics/restrictedAccess
dc.subjectOsteogenesis imperfectaen
dc.subjectQuality of lifeen
dc.subjectPsychologicalen
dc.subjectSocialen
dc.subjectPediatricen
dc.subjectChilden
dc.subjectAdulten
dc.subjectSystematic reviewen
dc.subjectKnowledge synthesisen
dc.subjectMixed-methodsen
dc.subjectWell-beingen
dc.titleThe psychosocial experience of individuals living with osteogenesis imperfecta: a mixed-methods systematic reviewen
dc.typeinfo:eu-repo/semantics/article
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