FAMILY ADAPTATION TO FRAGILE X SYNDROME ADOLESCENTS AND ADULTS

家庭对脆性 X 综合征青少年和成人的适应

• 批准号: 7482835
• 负责人: MARSHA RUTH MAILICK
• 金额: $ 31.76万
• 依托单位: UNIV OF NORTH CAROLINA CHAPEL HILL
• 依托单位国家: 美国
• 财政年份: 2008
• 资助国家: 美国
• 起止时间: 2008-07-01 至 2013-06-30
• 项目状态: 已结题
• 来源: https://reporter.nih.gov/project-details/7482835
• 关键词: Adaptive Behaviors; Adolescence; Adolescent; Adolescent Behavior; Adult; Age; Autistic Disorder; Behavior; Behavioral; Benchmarking; Biological; CGG repeat; Characteristics; Child; Child Behavior; Child Care; Child Development; Child Support; Daily; Data; Daughter; Development; Developmental Disabilities; Down Syndrome; Education; Environment; Expressed Emotion; FMR1; Family; Family Relationship; Fragile X Syndrome; Future; Genotype; Hand; Health; Home environment; Hydrocortisone; Income; Individual; Inherited; Intellectual functioning disability; Life; Longitudinal Studies; Maternal Health; Measures; Mental Health; Messenger RNA; Mothers; Nature; Numbers; Parent-Child Relations; Parenting behavior; Pathway interactions; Patient Self-Report; Pattern; Perception; Personal Satisfaction; Persons; Phenotype; Probability Samples; Problem behavior; Process; Public Health; Range; Rate; Reliance; Resources; Salivary; Sampling; Services; Severities; Shapes; Social support; Son; Source; Stress; Symptoms; Syndrome; System; Time; Variant; Well in self; autism spectrum disorder; caregiving; comparison group; design; diaries; disability; experience; high school; maternal stress; middle age; optimism; psychologic; response; self esteem; size; social; social stress; stressor; teenage mother

A. SPECIFIC AIMS Having a son or daughter with a developmental disability (DD) poses many challenges for families (Minnes, 1998). In most families, mothers assume the greatest share of child care responsibilities (Essex & Hong, 2005; Hastings, 2003; Heller et al., 1997). Consequently, a mother's experience of, and response to, those challenges are important determinants of her own health and well-being and of the quality of the caretaking context that she is able to create to support her child's development (Stoneman, 1997): As a group, mothers of individuals with fragile X syndrome (FXS) - the leading inherited cause of intellectual disability - fare rather poorly, displaying profiles of psychological well-being that are less like those of mothers of individuals with Down syndrome (DS) than mothers of individuals with autism (Abbeduto, Seltzer, Shattuck, Krauss, Orsmond, & Murphy, 2004). Mothers of individuals with FXS also have more pessimistic views about their child's future and less positive views of relationships and patterns of interaction within the family than mothers of individuals with DS, again being more similar to mothers of individuals with autism (Abbeduto et al., 2004; Lewis et al., 2006a). Nevertheless, there is considerable variability among mothers of individuals with FXS as regards their health, psychological well-being, and perceptions of the family (Bailey, Sideris, & Roberts, under review). Little is known, however, about the antecedents or consequences of these differences among mothers of individuals with FXS. Maternal well-being and perceptions of family relationships and adaptation are correlated with the severity of the challenging behaviors of the individual with FXS, including those behaviors reflective of autism, as well as with maternal characteristics (e.g., education) and contextual variables (e.g., number of children with FXS in the family and family income; Abbeduto et al., 2004). However, these relationships have been examined only for concurrently measured variables, making it impossible to determine the direction of effects (Esbensen, Seltzer, & Abbeduto, in press). Moreover, the pathways between maternal well-being and the family environment, on the one hand, and child behavior, on the other, likely involve a number of intervening processes that have not yet been investigated. How variations in biological vulnerability associated with maternal genotype (e.g., CCG repeat size, activation ratio, FMR1 mRNA levels) contribute to maternal wellbeing and responses to stress are unknown at this time as well (Esbensen et al., in press). Given the centrality of the family in the life of a person with FXS, the reliance of the service system on the family as the primary source of support, and the stress of lifelong family caregiving, there is an urgent public health and scientific need to understand the range of factors that influence the adaptation of individuals with FXS and the well-being of their families. The need for such data is particularly pressing for families whose son or daughter is in adolescence or young adulthood because these periods are associated with a decline in IQ, a slowing rate of adaptive behavior development, and an increase in severity of autistic-like behaviors for individuals with FXS (Hatton et al., 2006). Indeed, the evidence to date suggests that parenting stress and mental health challenges are greater for mothers parenting adolescents or adults with FXS than for mothers parenting children with FXS (Esbensen et al., in press). The proposed study, therefore, is designed to examine the longitudinal course and determinants of the well-being of the biological mothers of adolescents and adults with FXS, the nature of the daily stresses experienced by these mothers, and the family context that is both shaped by and shapes the individual with FXS. Moreover, the study will explore both variation among mothers of individuals with FXS and the extent to which such mothers differ from mothers parenting typically developing (TD) sons and daughters as well as mothers parenting sons and daughters with an autism spectrum disorder (ASD). Thus, these two comparison groups will provide useful benchmarks for gauging levels of maternal health and well-being and family adaptation for the FXS sample.

A.具体目标 有一个儿子或女儿患有发育障碍（DD）给家庭带来许多挑战（明尼苏达州， 1998）。在大多数家庭中，母亲承担最大的儿童保育责任（Essex＆Hong，2005年； 黑斯廷斯，2003年； Heller等，1997）。因此，母亲对那些的经历和回应 挑战是她自己的健康和福祉以及照料质量的重要决定因素 她能够创建以支持孩子的成长的背景（Stoneman，1997）：作为一个小组， 患有脆弱X综合征（FXS）的人 - 智力残疾的主要继承原因 - 票价而不是 不好，展示心理健康的概况，这些概况不像是 唐氏综合症（DS）比自闭症患者的母亲（Abbeduto，Seltzer，Shattuck，Krauss，Orsmond， ＆Murphy，2004年）。有FXS个人的母亲对孩子的未来也有更多的悲观观点 与个人母亲相比，家庭内部关系和互动模式的积极观点少 使用DS，再次与自闭症患者的母亲更相似（Abbeduto等，2004； Lewis等，，， 2006a）。然而，关于FX的个人的母亲在他们的 对家庭的健康，心理健康和看法（Bailey，Sideris和Roberts，正在审查中）。小的 但是，已经知道有关个人母亲之间这些差异的前提或后果 与FXS。孕产妇的福祉和对家庭关系和适应的看法与 通过FXS的个人挑战行为的严重性，包括反映自闭症的行为， 以及孕产妇特征（例如教育）和上下文变量（例如， 家庭和家庭收入中的FX； Abbeduto等，2004）。但是，这些关系已经 仅检查同时测量的变量，因此无法确定效果方向 （Esbensen，Seltzer和Abbeduto，印刷中）。此外，孕产妇福祉与 一方面，家庭环境和儿童行为另一方面可能涉及许多干预 尚未调查的过程。与生物学脆弱性的变化如何相关 母体基因型（例如CCG重复大小，激活比，FMR1 mRNA水平）有助于母体健康 目前对压力的响应也未知（Esbensen等人，印刷中）。 考虑到家庭生活中家庭生活中的中心地位，服务制度的依赖于 家庭是主要的支持来源，以及终身家庭护理的压力，有一个紧急的公众 健康和科学需求了解影响个人适应的因素范围 FXS和家人的福祉。对此类数据的需求特别是对儿子的家庭 或女儿处于青春期或成年年轻，因为这些时期与智商下降有关 自适应行为发展的速度放缓，以及自闭症行为的严重程度增加 患有FXS的个体（Hatton等，2006）。确实，迄今为止的证据表明，育儿压力和 母亲的育儿青少年或FXS的心理健康挑战比母亲更大 患有FXS的育儿儿童（Esbensen等人，印刷中）。因此，拟议的研究旨在检查 青少年和成人生物母亲的纵向过程和决定因素 使用FXS，这些母亲所经历的日常压力的本质以及两者都是家庭背景 由FXS塑造并塑造个人。此外，该研究将探索母亲之间的两种变化 FXS的个体以及此类母亲与母亲育儿的不同程度 （TD）儿子和女儿以及母亲的父母儿子和女儿患有自闭症谱系障碍 （ASD）。因此，这两个比较组将为衡量母体水平提供有用的基准 FXS样本的健康，福祉和家庭适应。