Psychological well-being of mothers of youth with fragile X syndrome: syndrome specificity and within-syndrome variability
Psychological well-being of mothers of youth with fragile X syndrome: syndrome specificity and within-syndrome variability
复制标题
DOI：
10.1111/j.1365-2788.2006.00907.x
复制
发表时间：
2006-12-01
影响因子：
3.6
通讯作者：
Orsmond, G.
中科院分区：
文献类型：
--
作者：
Lewis, P.;Abbeduto, L.;Orsmond, G.
背景关于父母幸福感的研究主要集中在唐氏综合征和自闭症;然而，与其他诊断条件相比，脆性X综合征可能会给父母带来不同的挑战。此外，患有脆性X综合征的年轻人之间存在相当大的差异;例如，25%至33%的受影响青年符合自闭症共病诊断的标准。患有脆性X综合征的年轻人的父母可能会经历不同程度和模式的压力，这取决于他们的后代是否患有自闭症的共病诊断。在目前的研究中，我们比较了三组年轻男性的母亲在心理健康和压力方面的措施：那些患有脆性X综合征和自闭症共病诊断的母亲;那些单独患有脆性X综合征的母亲;方法样本由患有脆性X综合征和共病自闭症的青少年和年轻成年男性的母亲组成（n = 9），脆性X综合征（n = 19）和唐氏综合征（n = 19）。我们使用自闭症行为检查表（Autism Behavior Checklist）对所有青少年进行了自闭症筛查，该检查表由母亲、父亲和教师完成，得分高于建议临界值的青少年由有执照的心理学家进行评估，以确定自闭症状态。这三组青年在实际年龄（分别为16.4、15.8和16.0岁）或非语言心理年龄（分别为3.8、3.9和3.8岁）方面没有差异。母亲们完成了几项自我报告措施。这些措施评估了目前的心理健康状况（例如流行病学研究中心抑郁量表），对儿子和家庭功能的看法（例如，积极情感指数，它测量母亲对儿子的亲密感，以及儿子对母亲的相互亲密感，如母亲所感知的），和方法来应对他们的儿子的残疾[例如，多维应对量表（科普），其中措施的情绪为重点和解决问题为重点的应对]。结果结果表明，脆性X综合征创造更多的挑战，母亲的心理健康比唐氏综合征，脆性X综合征和自闭症的结合可能特别具有挑战性。然而，群体之间的差异主要表现为对受影响的儿子和家庭内部关系的关注，而不是心理健康水平较低。因此，患有脆性X综合征的儿子的母亲，无论儿子的自闭症状况如何，都比患有唐氏综合征的儿子的母亲对儿子的未来更加悲观，家庭内部的冲突也更多。此外，与脆性X综合征和共病自闭症的儿子的母亲报告较低水平的互惠亲密比其他两组的mothers.Conclusion，我们认为可能的原因，这些产妇的差异，对临床实践的影响，需要为今后的研究，了解儿童和上下文因素的重要性，以及导致这些差异的动态。
Background Research on parental well-being has focused largely on Down syndrome and autism; however, fragile X syndrome is likely to pose different challenges for parents compared with these other diagnostic conditions. Moreover, there is considerable variability among youth with fragile X syndrome; for example, 25% to 33% of affected youth meet criteria for a co-morbid diagnosis of autism. It is likely that parents of youth with fragile X syndrome will experience different degrees and patterns of stress, depending on whether their offspring do or do not have a co-morbid diagnosis of autism. In the present study, we compared mothers of three groups of young males on measures of psychological well-being and stress: those with fragile X syndrome and a co-morbid diagnosis of autism; those with fragile X syndrome alone; and those with Down syndrome.Method The sample consisted of mothers of adolescent and young adult males with fragile X syndrome and co-morbid autism (n = 9), fragile X syndrome alone (n = 19), and Down syndrome (n = 19). We screened all youth for autism using the Autism Behavior Checklist, which was completed by mothers, fathers and teachers, and the youth who scored above the suggested cut-off were evaluated by a licensed psychologist to determine autism status. The three groups of youth did not differ in chronological age (16.4, 15.8 and 16.0 years, respectively) or non-verbal mental age (3.8, 3.9 and 3.8 years, respectively). Several self-report measures were completed by mothers. These measures assessed current mental health status (e.g. the Center for Epidemiological Studies Depression Scale), perceptions of their son's and family's functioning (e.g. the Positive Affect Index, which measures closeness felt by the mother to her son and also reciprocated closeness felt by the son towards the mother, as perceived by the mother), and approach to coping with their son's disability [e.g. the Multidimensional Coping Inventory (COPE), which measures emotion-focused and problem-solving focused coping].Results The results suggest that fragile X syndrome creates more challenges to maternal psychological well-being than Down syndrome, and that the combination of fragile X syndrome and autism can be particularly challenging. Differences among groups, however, were manifested mainly as concerns about the affected son and about relationships within the family rather than as lower levels of mental health. Thus, mothers of sons with fragile X syndrome, regardless of the son's autism status, reported more pessimism about the son's future and more conflict within the family than mothers of sons with Down syndrome. Additionally, mothers of sons with fragile X syndrome and co-morbid autism reported lower levels of reciprocated closeness than the other two groups of mothers.Conclusion We consider possible causes of these maternal differences, the implications for clinical practice, needs for future research, and the importance of understanding child and contextual factors as well as the dynamics leading to these differences.