Families of those with PWS experience a great deal of stress, particularly during adolescence. In this study, we are funding the development of a new behavioral intervention, aimed at developing more effective coping skills in fathers of adolescents with PWS, as a means to improve overall family well being. Dr. Forster will work with fathers of adolescents with PWS using an established program to teach healthy coping skills. The goal is for father to be less stressed and more engaged, thereby improving the overall functioning and well being of the entire family.
Theresa Strong, Director of Research Programs, shares details on this project in this short video clip.
Watch the full webinar describing the 11 research projects funded in this grant cycle here.
Families of people with PWS experience more stress than families of people with any other developmental disability, especially during the adolescent and young adult years. Stress is the highest in mothers followed by fathers. It has been difficult to engage mothers in formal stress reduction interventions, perhaps because they are overwhelmed with their care-giving demands. In this study, we propose to enroll fathers of intact families with an adolescent with PWS age 12-18 years in a psychological intervention delivered via a series of on-line groups. We believe that fathers will be accessible and amenable to this technological approach. Acceptance and Commitment Training (ACT) has been used successfully with families of children with autism and other disabilities in over 100 studies. This is the first time ACT will be applied to PWS. Through a series of mindfulness exercises, ACT teaches cognitive flexibility: the ability to focus on the here-and-now while becoming aware of inner thoughts and emotions that set up road blocks and interfere with the ability to achieve goals consistent with personal values. The ultimate objective of this flexibility is to move toward a rewarding life style with healthy coping strategies and away from distress and avoidance, despite difficult circumstances. We believe that by modifying the father’s perception of stress, he will be better able to devote time, energy and emotional support to the family in order to help stabilize the home environment. As a secondary target, we hope to see an improved family environment resulting in decreased stress and increased parenting satisfaction for the mother. Fathers will be recruited through advertisements on FPWR, PWSA and chapter websites. Family demographics, home environment, and the adolescent’s degree of behavioral difficulty will be assessed at the outset. Then, we will administer a series of questionnaires measuring stress and cognitive flexibility in fathers before, during, and after the intervention. Maternal stress, parenting satisfaction and home environment measures will be completed before and after the intervention. At the conclusion of this intervention, we hope to see that fathers will be less stressed and more engaged in family function to ease family stress and decrease emotional distress for the mother. Although this research addresses family function rather than the clinical care of the individual with PWS, it has the potential to improve the adolescent’s behavior in two ways. First, by improving family function, parents may be more able to manage the home and family environment resulting in decreased stress for everyone. Second, decreased family stress may increase the likelihood that family members will participate more fully with behavioral treatment recommendations. Next steps: Based upon the success of this pilot, we plan to develop similar on-line programs for mothers and siblings of adolescents with PWS. Also, we would like to make this family-focused intervention available to parents of children at other developmental levels, and offer “ACT for PWS” training to staff of PWS multidisciplinary clinics across the USA and Canada.
Research Outcomes: Public Summary
Acceptance and Commitment Training (ACT) is a mindfulness intervention that increases cognitive flexibility and teaches coping strategies to expand the quality-of-life experience. Using an ACT protocol of 4 sessions delivered online via ZOOM twice monthly to a group of fathers of adolescents with PWS, we expected to see increased engagement of fathers in the family process and reduced family stress as measured by changes in scores from baseline on several questionnaires: the PSS-10 (the Cohen Perceived Stress Scale), the FES (Family Environment Scales) and the 6-PAQ (an ACT-based instrument measuring perception of parenting skills). Additionally, the study group was described by the Zarit-Burden Interview (demographics) and the PWSBQ (the Prader-Willi Syndrome Behavior Questionnaire).
The online format for the data collection and 4-session ACT protocol performed well. The questionnaires that were selected for this study (PWSBQ, Zarit-Burden Interview, PSS-10, FES, 6-PAQ) were informative and had good face validity. The greatest challenge was recruitment; we expected 20 families. Our study group consisted of 4 fathers of adolescents with PWS who resided in intact families. Demographic measures were very similar for all parents participating with respect to age and level of education; amount of exercise, participation in leisure activities, and sleep duration were more variable both among and between mothers and fathers. The 4 adolescents ranged in age from 14-17 years; there were 2 males, 2 females, 2 with deletion subtype and 2 with maternal uniparental disomy. Despite gender and genetic subtype differences, behavioral responses on the PWSBQ were similar. Higher scores were reported on the rigid/inflexibility domain followed by food-related behaviors, and both these domains showed the largest difference in parent’s scores, e.g., father’s scores were consistently higher than the mothers except for the social interaction domain, which was marginally different and the lowest scoring domain. Food-seeking behaviors and physical symptoms, such as skin-picking, were not as highly related to stress and burden as were rigidity and issues with discipline and anger. On the PSS-10, father’s stress levels decreased over time concurrent with the 4 ACT sessions and increased a bit after the follow up interval, an expected finding. Data from the 6-PAQ (an ACT-based questionnaire) was more divergent initially and converged at the end, suggesting that discussions during ACT sessions may have normalized father’s perceptions of their parenting role. In particular, “acceptance of difficult behavior” and “awareness of not being present” increased over time. The FES data showed that family conflict decreased after the 4 ACT sessions from both parents’ perspectives.
In summary, measurements of behaviors typical for adolescents with PWS were similar among the 4 adolescents in this study, and fathers consistently rated those behaviors as more problematic than the mothers. Fathers level of stress was scored higher than the index score in all cases and more than twice as high in 3 of 4 subjects. We found that stress scores decreased across the 4 ACT sessions and rebounded a little after the post intervention interval, suggesting that stress or perception of stress decreased with ACT. We also found that father’s perception of their child’s behavior and their parenting role changed in a positive direction with ACT. Finally, perception of family conflict decreased concurrent with ACT intervention. Therefore, there is evidence to suggest that ACT intervention delivered across 4 sessions via ZOOM was both feasible and effective to decrease perception of stress, family conflict, and increase parenting satisfaction.
Jan Forster, MD
Jan Forster, MD