{"title":"22q11.2缺失综合征患儿家庭适应能力的抗逆性因素:家长视角的混合方法研究。","authors":"Maria Caples, Bridie McCarthy, Eileen Savage","doi":"10.1177/17446295231186851","DOIUrl":null,"url":null,"abstract":"<p><p>22q11.2 deletion syndrome is a rare multisystem genetic disorder with over 200 associated characteristics, occurring in various combinations and severity. Extensive biomedical research has been undertaken on 22q11.2 deletion syndrome, however, there is a dearth of research on families' experiences of managing a family member with this condition. The complex and at times serious phenotypical presentation of the syndrome can make the management of the condition difficult for families. The aim of this mixed method explanatory sequential study was to investigate family hardiness as a resilience factor for adaptation in families of children with 22q11.2 deletion syndrome from parents' perspectives. We found that adaptation scores increased by 0.57 points (95% CI: 0.19-0.94) for every one-point increase in family hardiness score. Qualitative results indicated that acceptance of the child's diagnosis and support positively influenced hardiness whereas fears about the future and their experiences of loss negatively influenced hardiness.</p>","PeriodicalId":46904,"journal":{"name":"Journal of Intellectual Disabilities","volume":" ","pages":"738-758"},"PeriodicalIF":1.5000,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11465608/pdf/","citationCount":"0","resultStr":"{\"title\":\"Hardiness as a Resilience Factor for Adaptation in Families of Children With 22q11.2 Deletion Syndrome: A Mixed Methods Study of Parents' Perspectives.\",\"authors\":\"Maria Caples, Bridie McCarthy, Eileen Savage\",\"doi\":\"10.1177/17446295231186851\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>22q11.2 deletion syndrome is a rare multisystem genetic disorder with over 200 associated characteristics, occurring in various combinations and severity. Extensive biomedical research has been undertaken on 22q11.2 deletion syndrome, however, there is a dearth of research on families' experiences of managing a family member with this condition. The complex and at times serious phenotypical presentation of the syndrome can make the management of the condition difficult for families. The aim of this mixed method explanatory sequential study was to investigate family hardiness as a resilience factor for adaptation in families of children with 22q11.2 deletion syndrome from parents' perspectives. We found that adaptation scores increased by 0.57 points (95% CI: 0.19-0.94) for every one-point increase in family hardiness score. Qualitative results indicated that acceptance of the child's diagnosis and support positively influenced hardiness whereas fears about the future and their experiences of loss negatively influenced hardiness.</p>\",\"PeriodicalId\":46904,\"journal\":{\"name\":\"Journal of Intellectual Disabilities\",\"volume\":\" \",\"pages\":\"738-758\"},\"PeriodicalIF\":1.5000,\"publicationDate\":\"2024-09-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11465608/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of Intellectual Disabilities\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1177/17446295231186851\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2023/7/4 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q2\",\"JCRName\":\"EDUCATION, SPECIAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Intellectual Disabilities","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1177/17446295231186851","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2023/7/4 0:00:00","PubModel":"Epub","JCR":"Q2","JCRName":"EDUCATION, SPECIAL","Score":null,"Total":0}
Hardiness as a Resilience Factor for Adaptation in Families of Children With 22q11.2 Deletion Syndrome: A Mixed Methods Study of Parents' Perspectives.
22q11.2 deletion syndrome is a rare multisystem genetic disorder with over 200 associated characteristics, occurring in various combinations and severity. Extensive biomedical research has been undertaken on 22q11.2 deletion syndrome, however, there is a dearth of research on families' experiences of managing a family member with this condition. The complex and at times serious phenotypical presentation of the syndrome can make the management of the condition difficult for families. The aim of this mixed method explanatory sequential study was to investigate family hardiness as a resilience factor for adaptation in families of children with 22q11.2 deletion syndrome from parents' perspectives. We found that adaptation scores increased by 0.57 points (95% CI: 0.19-0.94) for every one-point increase in family hardiness score. Qualitative results indicated that acceptance of the child's diagnosis and support positively influenced hardiness whereas fears about the future and their experiences of loss negatively influenced hardiness.
期刊介绍:
The principal aim of the journal is to provide a medium for the exchange of best practice, knowledge and research between academic and professional disciplines from education, social and health settings to bring about advancement of services for people with intellectual disabilities. The idea of a practice-led journal is both exciting and timely. This journal serves as a medium for all those involved with people with intellectual disabilities to submit and publish papers on issues relevant to promoting services for people with intellectual disabilities.