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

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

Author Tsimicalis, Argerie Google Scholar
Denis-Larocque, Gabrielle Google Scholar
Michalovic, Alisha Google Scholar
Lepage, Carolann Google Scholar
Williams, Karl Google Scholar
Yao, Tian-Ran Google Scholar
Palomo, Telma Autor UNIFESP Google Scholar
Dahan-Oliel, Noemi Google Scholar
Le May, Sylvie Google Scholar
Rauch, Frank Google Scholar
Abstract Osteogenesis 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 = 800

comprising 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.
Keywords Osteogenesis imperfecta
Quality of life
Psychological
Social
Pediatric
Child
Adult
Systematic review
Knowledge synthesis
Mixed-methods
Well-being
xmlui.dri2xhtml.METS-1.0.item-coverage Dordrecht
Language English
Sponsor Newton Foundation
Tunis Shriners
Quebec Nursing Intervention Research Network
Date 2016
Published in Quality Of Life Research. Dordrecht, v. 25, n. 8, p. 1877-1896, 2016.
ISSN 0962-9343 (Sherpa/Romeo, impact factor)
Publisher Springer
Extent 1877-1896
Origin http://dx.doi.org/10.1007/s11136-016-1247-0
Access rights Closed access
Type Article
Web of Science ID WOS:000380005900002
URI https://repositorio.unifesp.br/handle/11600/57498

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