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Exploring the impact of Osteogenesis Imperfecta on families: A mixed-methods systematic review

Hill, M; Lewis, C; Riddington, M; Crowe, B; DeVile, C; Gotherstrom, C; Chitty, L; (2019) Exploring the impact of Osteogenesis Imperfecta on families: A mixed-methods systematic review. Disability and Health Journal , 12 (3) pp. 340-349. 10.1016/j.dhjo.2018.12.003. Green open access

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Abstract

Background: Osteogenesis Imperfecta (OI) is a rare genetic condition whose key characteristic is increased bone fragility. OI has the potential to impact upon all family members, making it important to consider the challenges families face, how they cope and their support needs as the affected individual moves from childhood through to adult life. / Objective: To conduct a mixed-methods systematic review investigating the experiences of families when a family member is affected with OI. / Methods: A systematic search of seven electronic databases, relevant patient organisation websites and reference lists was conducted. Data extraction was performed for all studies that met the eligibility and quality criteria. Results were synthesised following the principles of thematic analysis. / Results: One mixed-method, six qualitative and six quantitative studies were included in the review. Three overarching themes were identified through thematic analysis: Impact of OI on the psychosocial wellbeing of families, impact on family life and evolving roles and relationships. Fear of fractures and the uncertainty of when the next fracture will occur are key issues that permeate all areas of family life and impact upon all family members. / Conclusion: The experiences, coping strategies and support needs of families affected by OI were highly variable and changed over time. Future research should address the need for adaptive health and education interventions that support all family members.

Type: Article
Title: Exploring the impact of Osteogenesis Imperfecta on families: A mixed-methods systematic review
Open access status: An open access version is available from UCL Discovery
DOI: 10.1016/j.dhjo.2018.12.003
Publisher version: https://doi.org/10.1016/j.dhjo.2018.12.003
Language: English
Additional information: Copyright © 2019 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Keywords: Osteogenesis Imperfecta, Family, Mixed-methods systematic review, Psychosocial
UCL classification: UCL
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health > Genetics and Genomic Medicine Dept
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health > Population, Policy and Practice Dept
URI: https://discovery.ucl.ac.uk/id/eprint/10089718
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