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Adaptation and resilience in families of children with 22q11.2 deletion syndrome: a mixed methods study
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Date
2019-11-19
Authors
Caples, Maria
Journal Title
Journal ISSN
Volume Title
Publisher
University College Cork
Published Version
Abstract
Background: 22q11.2 deletion syndrome is a rare multisystem genetic disorder with over 200 associated characteristics, occurring in various combinations and severity, with an estimated incidence of 1 in 4,000 live births. The phenotype of syndrome is much varied, with some people more seriously affected then others. In addition, a rare disease can bring a range of challenges for those affected and their families which can result in people becoming marginalised psychologically, socially, culturally and economically. Extensive biomedical research has been undertaken on 22q11.2 deletion syndrome, however, there is a dearth of research on families’ experiences.
Study Aim: To investigate the relationship between resilience factors and adaptation in families of children with 22q11.2 deletion syndrome.
Methods: An explanatory sequential mixed methods design was used to investigate adaptation and resilience in families of children with 22q11.2 deletion syndrome. Families participated in a survey (n=64) and qualitative interviews (n=9). Quantitative data were analysed using IBM SPSS Statistics (Version 18) and qualitative data were analysed using content analysis.
Results:Three variables that best explained adaptation in families of children with 22q11.2 deletion syndrome were identified: family hardiness, condition management effort, and condition management ability. Adaptation scores increased by 0.57 points (95% CI: 0.19 to 0.94) for every one-point increase in family hardiness score, by 0.95 (95% CI: 0.33 to 1.56) for every one-point increase in condition management ability score and decreased by 0.84 (95% CI: -1.55 to -0.13) for every one-point increase in effort scores.
The qualitative data provided additional insights to the independent variables that were found to be significantly associated with adaptation for families of children with 22q11.2 deletion syndrome. These variables were condition management effort, condition management ability and family hardiness.
Conclusion: This study provides insight to adaptation and resilience specific to families of children with 22q11.2 deletion syndrome. A key implication of the findings is the need for a family centered, integrated approach to care for families of children with 22q11.2 deletion syndrome.
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Keywords
Adaptation , Resilience , Families , 22q11.2 deletion syndrome
Citation
Caples, M. 2020. Adaptation and resilience in families of children with 22q11.2 deletion syndrome: a mixed methods study. PhD Thesis, University College Cork.