Qualitative Differences across Genetic Subtypes of Prader-Willi Syndrome: Efficacy of a Remote Play-Based Parent Training

• In Event: Using computer technology to expand detection and intervention for children with developmental disorders

Abstract

Introduction: Preliminary research demonstrates that children with Prader-Willi Syndrome (PWS) have impaired pretend play abilities and parent-child interactions during play, which may be markers of larger social cognitive deficits characteristic of this disorder (Dimitropoulos et al., 2019; Zyga and Dimitropoulos, 2020). Targeting skill building through play and parent-child interactions in children may strengthen adaptive skills, socioemotional understanding, and the ability to engage in and appropriately interact in social situations while also decreasing rigid and repetitive behaviors (Russ, 2014). In addition, developing behavioral interventions via telehealth allows for increased access for individuals with PWS, who are often faced with significant barriers such as distance and cost of in-person trials (Vismara et al., 2013). The present study reports findings from our ongoing examination of the Play-based Remote Enrichment To ENhance Development (PRETEND) parent training program for parents of preschoolers with PWS.

Method: The current study evaluated the efficacy of the PRETEND-Preschool parent training program, an 8-week remotely delivered program that includes once weekly sessions, 45-60 minutes in length for parents of children aged 3-5 years. 18 participants completed both in-person baseline and post-intervention visits, where measures were used to directly assess children’s pretend play skills and cognitive skills, and parent-child interactions in play. Additionally, parents completed parent report measures of children’s social cognitive skills, social skills, and measures of their own stress related to their children. During the intervention period, parents randomized to the intervention group (INV) worked directly with the interventionist via videoconferencing software to complete an adapted play-based program aimed at increasing imagination and emotional expression in typically developing children (Moore & Russ, 2008). The PRETEND-Preschool program consists of 1) individual parent sessions to review play and related cognitive, emotional and behavioral skills, 2) live coaching-play sessions in which the interventionist instructs the parent remotely during their play with the child, and 3) recorded play sessions in which the parent and child play independently together.

Results: No significant interactions were observed between the INV and WC (waitlist control) groups pre to post intervention on any of the APS-P variables. Given the small sample size, a series of qualitative analyses were applied to the data to further examine the differences in change between the INV and WC participants within the two genetic subtypes (UPD and DEL) of PWS. Results indicated that the intervention participants with UPD demonstrated meaningful improvement above and beyond that of the waitlist control participants on multiple play variables including increasing Imagination, Organization, Symbolic play, and Affect frequency. The DEL subgroup demonstrated fewer improvements across play variables.

Discussion: These results are the first to support preliminary efficacy of this intervention in preschool-aged children with PWS. Findings indicate that the remote play-based intervention significantly improved cognitive and affective play skills, and amount of time spent in symbolic play, specifically for children in the UPD subgroup who underwent the intervention. Results from this study indicate an important new avenue of feasible and accessible behavioral intervention for preschool-aged children with PWS, and specific subgroup effects will be further discussed.

Authors

• Anastasia Dimitropoulos, Case Western Reserve University
  Presenting Author

• Ellen Doernberg, Case Western Reserve University
  Non-Presenting Author

• Sandra Russ, Case Western Reserve University
  Non-Presenting Author