What about adolescence? Living with genetic risk

描述(由申请人提供)： 了解学习遗传风险信息给儿童带来的后果主要是基于从成年人那里推断出来的数据。还没有进行过评估遗传风险知识与儿童自我概念之间关系的研究。我们也尚未了解儿童如何应对遗传信息，以及他们的应对策略如何受到父母应对方法的影响。本研究的目的是描述青春期女孩的自我概念、应对和适应与X连锁疾病脆性X的遗传风险知识之间的关系。我们还将描述母亲使用的策略来帮助她们的女儿应对这些信息。我们将使用多组队列设计，研究60名女孩(15-18岁)及其母亲，他们来自脆性X基因家庭。我们将招募母女二人组，了解女孩的遗传风险：1)携带者，2)非携带者，3)有风险的携带者。每一位被染色的母女都将完成一些定量和定性的测量。我们的具体目标是：1)描述青春期女孩的自我概念、应对和适应，了解她们患X连锁疾病的遗传风险(携带者、非携带者和高危人群)。2)描述a)母亲如何选择与女儿讨论遗传风险信息，b)父母对应对行为的“指导”，以及c)她们对女儿如何管理这些信息的看法。3)描述青春期女孩对脆性X的知识及其遗传方式；她们如何了解自己的携带者、非携带者或高危状态；她们对携带者、非携带者和高危等词的定义；以及她们认为遗传风险知识如何影响家庭和同龄人关系。4)建立具有这三类遗传风险知识的青春期女孩队列的基线测量，以便我们可以在未来的纵向研究中跟踪发展中的自我概念，考虑发展任务、应对行为、适应和父母角色实验。我们假设：a.携带者和高危青春期女孩在自我概念、应对和适应方面比非携带者更相似；b.高度重视未来父母角色的携带者和高危青春期女孩的自我概念低于那些不看重未来父母角色的女孩。

DESCRIPTION (provided by applicant): Understanding of the consequences to children of learning genetic risk information is based primarily on data extrapolated from adults. No studies have been done that assess the relationship between knowledge of genetic risk and a child's self-concept. We also have yet to learn how children cope with genetic information and how their coping strategies are affected by their parents' approach to coping. The purpose of this study is to describe the relationship among adolescent girl's self-concept, coping, and adjustment associated with knowledge of genetic risk of an X-linked disorder, fragile X. We will also describe strategies mothers use to help their daughters cope with this information. Using a multigroup cohort design, we will study 60 girls (15- 18 yrs) and their mothers from families with fragile X. We will enroll mother/daughter dyads with 3 categories of knowledge about the girl's genetic risk: 1) carrier, 2) noncarrier, and 3) at-risk to be a carrier. Each mother/daughter dyed will complete a number of quantitative and qualitative measures. Our specific aims are to: 1) Describe self-concept, coping, and adjustment in adolescent girls with knowledge of their genetic risk (carrier, noncarrier, and at-risk) for an X-linked disorder. 2) To describe a) how mothers have chosen to discuss genetic risk information with their daughters, b) parental "coaching" of coping behaviors, and c) their perceptions of how their daughters are managing this information. 3) Describe adolescent girls' knowledge of fragile X and how it is inherited; how they learned their carrier, noncarrier, or at-risk status; their own definitions of the words carrier, noncarrier, and at-risk; and how they think knowledge of genetic risk influences family and peer relationships. 4) To establish baseline measurements on a cohort of adolescent girls with these 3 categories of knowledge of genetic risk so that we can follow the developing self-concept in future longitudinal studies considering developmental tasks, coping behaviors, adjustment, and parental role experimentation. We hypothesize that: a. Carrier and at-risk adolescent girls will be more similar than noncarriers in self-concept, coping, and adjustment, b. Carrier and at-risk adolescent girls who place a high value on a future parental role will have a lower self-concept than those that do not.