FAMILY ADAPTATION TO FRAGILE X SYNDROME ADOLESCENTS AND ADULTS

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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.