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。

IF 1.5 4区 医学 Q2 EDUCATION, SPECIAL Journal of Intellectual Disabilities Pub Date : 2024-09-01 Epub Date: 2023-07-04 DOI:10.1177/17446295231186851
Maria Caples, Bridie McCarthy, Eileen Savage
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引用次数: 0

摘要

22q11.2 缺失综合征是一种罕见的多系统遗传疾病,有 200 多种相关特征,以不同的组合和严重程度出现。有关 22q11.2 缺失综合征的生物医学研究已广泛开展,然而,有关家庭成员管理该疾病的经验的研究却十分匮乏。22q11.2 缺失综合征的表型复杂,有时甚至很严重,这给家庭的病情管理带来了困难。这项混合方法的解释性顺序研究旨在从父母的视角出发,调查家庭坚韧性作为 22q11.2 缺失综合征患儿家庭适应能力的一个弹性因素。我们发现,家庭坚韧性得分每增加一分,适应性得分就会增加 0.57 分(95% CI:0.19-0.94)。定性结果表明,接受孩子的诊断和支持对适应性有积极影响,而对未来的恐惧和失去孩子的经历则对适应性有消极影响。
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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.

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来源期刊
CiteScore
3.40
自引率
13.30%
发文量
82
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.
期刊最新文献
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