For Year 1, our project aims were: 1) to characterize the social, cognitive, and affective processes in preschoolers with PWS (by genetic subtype), in comparison to preschoolers with ASD and typically developing children, and 2) to pilot a remotely delivered parent education program to determine if it would be feasible and effective for families with young children with PWS in increasing play, social understanding, and decreasing problem behaviors.
Thus far, results of the study indicate the following: 1. Remote delivery of parent training is feasible in this population, and may be an effective option for helping with behavior, emotion, and social functioning in preschoolers with PWS; 2. Measures taken by both researchers and parents indicate that even at the preschool level, children with mUPD have greater social-cognitive challenges compared with children with pDEL. 3. The PRETEND parent training program could help to improve quality of parent-child interactions and could decrease problem behaviors. Additional recent findings from the Dimitropoulos Lab show that direct remote intervention with school-age children with PWS may increase emotional expression, quality of imaginative play, and overall social cognitive skills, while decreasing challenging behavior. Taken together these findings indicate that a combination of remotely delivered parent-training and direct behavioral intervention could potentially yield positive emotional, social, and behavioral results for preschool and school-age children with PWS.
Therefore, the Year 2 Aims are as follows. In Aim 1, we will evaluate the efficacy of a combined parent training and direct child intervention program (PRETEND: Play-based Remote Enrichment To ENhance Development) via remote delivery for children with PWS and their parents. 40 children with PWS (ages 3-9) will be recruited, randomizing 20 children to immediate intervention (10 preschool, 10 school age), and 20 to a waitlist control group (10 preschool, 10 school-age). The program will be tailored specifically to the needs of each age group (i.e., more parent training for families of preschoolers, more direct intervention for school age children). Overall, we predict that this combined program will be more effective at increasing social cognitive ability and decreasing challenging behavior for both preschool and school-age children with PWS. In Aim 2, we will evaluate the use of eye-tracking as a potential indicator of social attention in PWS. All participants will complete tasks that measure social cognitive ability and undergo a passive eye-tracking task to evaluate social attention both at the baseline and post-intervention visits. As our previous findings suggest, social cognitive challenges may be present in PWS as early as the preschool years, thus it is important to also understand how these difficulties relate to factors such as looking time or gaze fixation to social cues and how this may also be impacted by behavioral intervention.
*Funded by FPWR-Canada
Research Outcomes: Publications
Show me what happens next: Preliminary efficacy of a remote play-based intervention for children with Prader-Willi syndrome. Dimitropoulos A, Zyga O, Doernberg E, Russ SW. Res Dev Disabil. 2021 Jan;108:103820. doi: 10.1016/j.ridd.2020.103820. Epub 2020 Dec 9.PMID: 33307337
Early Social Cognitive Ability in Preschoolers with Prader-Willi Syndrome and Autism Spectrum Disorder. Dimitropoulos A, Zyga O, Russ SW.J Autism Dev Disord. 2019 Nov;49(11):4441-4454. doi: 10.1007/s10803-019-04152-4. PMID: 31388797
The PRETEND Program: Evaluating the Feasibility of a Remote Parent-Training Intervention for Children with Prader-Willi Syndrome. Zyga, O., Russ, S., & Dimitropoulos, A. (2018). American Journal on Intellectual and Developmental Disabilities, 123(6), 574-584
Evaluating the feasibility of a play-based telehealth intervention program for children with Prader-Willi syndrome. Dimitropoulos, A., Zyga, O, & Russ, S. (2017). Journal of Autism and Developmental Disorders, 47(9), 2814-2825.
Anastasia Dimitropoulos, PhD
$77,679 (This Project was funded by the Foundation for Prader-Willi Research Canada.
Case Western Reserve University