PWS Smart-Start – A Randomized Clinical Trial
Funding Summary
This project aims to evaluate a parent training program (PWS Smart Start) for helping caregivers develop the skills they need to address challenging behavior and skill deficits common among children with Prader-Willi Syndrome. This project begins to lay the foundation for behavior analytic services for children with PWS, with the end goal of demonstrating the effectiveness of PWS Smart-Start specifically and, in general, the value of behavior analytic services that can be shared with key stakeholders for a variety of purposes, including promotion of insurance coverage for ABA.
Dr. Theresa Strong, Director of Research Programs, shares details on this project in this short video clip.
Lay Abstract
This project aims to evaluate a parent training program (PWS Smart Start) for helping caregivers develop the skills they need to address challenging behavior and skill deficits common among children with Prader-Willi Syndrome. This study will introduce 80 families (two groups of 40 caregivers) to the parent training program, and evaluate the outcomes on caregiver learning, child behavior, and the health, wellness, and stress levels of children and caregivers. PWS-Smart Start was developed using the science of Applied Behavior Analysis (ABA). Evidence-based behavior analytic practice has been shown to be very effective in helping parents of children with other developmental disabilities acquire the skills they need to help their children; however, very little work has been done to extend this intervention to children with PWS and their families. Due to the rare nature of PWS and the lack of effective interventions, parents, and caregivers are often left with insufficient strategies for handling their child’s behavior. It is imperative to develop behavioral interventions to support caregivers by reducing caregiver burden and burnout and to help children develop the skills necessary to succeed socially and academically. This project begins to lay the foundation for behavior analytic services for children with PWS, with the end goal of demonstrating the effectiveness of PWS Smart-Start specifically and, in general, the value of behavior analytic services that can be shared with key stakeholders for a variety of purposes, including promotion of insurance coverage for ABA. Additionally, this intervention has the potential for international usefulness as it can be delivered worldwide using remote technologies.
Research Outcome
PWS Smart Start showed promise as a way to provide families with practical behavioral support tailored to Prader-Willi syndrome (PWS). In this study, caregivers who received the program reported better positive-parenting practices and fewer food-related and non-food repetitive behaviors than caregivers who were still waiting to begin, after differences in starting scores were taken into account. Caregivers also rated the program highly. The findings provide preliminary evidence of benefits in these specific areas and identify priorities for improving longer-term support.
Children with PWS may experience emotional outbursts, repetitive behavior, skin picking, difficulty with changes in routine, and food-related behavioral concerns. These challenges can affect learning, everyday activities, and family life. Caregivers need strategies that account for PWS, yet many families have difficulty finding professionals with relevant expertise. Among caregivers who completed this study, 86% reported that they had never received caregiver training before joining the program. All agreed at the beginning of the study that families need behavioral support tailored to PWS.
PWS Smart Start was delivered through 16 weekly, individual online sessions with a Board-Certified Behavior Analyst. Families participated from their homes in the United States and internationally. Caregivers learned how to understand and track behavior, prevent predictable difficulties, teach communication and other useful skills, and reinforce appropriate behavior. The program also addressed schedules, waiting, self-calming, food security, and ways to use skills across daily routines. Caregivers practiced the strategies, completed activities between sessions, and worked with their coach to develop a plan tailored to their child's strengths and needs.
Sixty-five caregiver-child pairs were randomly assigned to receive the program immediately or to continue their usual services while waiting. Fifty-eight pairs completed all study assessments and were included in the analyses. Children were 3-12 years old. The second group then received PWS Smart Start, allowing researchers to examine changes following participation in both groups. The first group also completed a later assessment to examine whether changes persisted after coaching ended.
Researchers compared each family's reports before and after its training period. These analyses showed a wider range of encouraging changes. Caregivers reported reductions in food-related behavior, repetitive behavior, skin picking, rigidity, disruptive behavior, and the overall interference associated with behavioral symptoms. They also reported less distress in several areas of parenting, lower burnout, greater use of positive and behavior-based parenting strategies, and a modest improvement in family quality of life. Total parenting stress did not change significantly. These before-and-after changes do not establish that the program alone caused the improvements. In the randomized comparison, differences in stress, burnout, family quality of life, and several child-behavior outcomes did not reach statistical significance.
The study also examined whether the program could be delivered consistently online. Coaches completed an average of 99.7% of the required program components. Caregiver-acquisition checklists, which assessed completion of training components and homework, averaged 99.6%. Independent reviewers checked more than one-third of sessions and showed high agreement with the original ratings. These results support the feasibility of providing structured, individualized caregiver training through telehealth. They do not establish that every strategy was used independently or consistently in all daily situations, which remains an important question for future research.
Caregiver feedback was strongly positive. Forty-eight caregivers completed the satisfaction survey. All respondents agreed that the content was relevant, important, and tailored to PWS, and all would recommend the program to other families. About 96% thought the number of sessions was appropriate. Among those answering the relevant questions, all reported learning new skills and feeling better able to help their child. Most reported changing how they arranged the home environment. About 94% wanted continued access to their coach after the program ended, indicating that families valued the support and wanted help applying it over time.
Children's responses varied. Eight caregivers completed frequent reports of individualized behaviors. Four of five children with emotional-dysregulation targets improved after caregivers completed the emotional-regulation module, although behavior remained variable and the study did not independently test that module. Other concerns persisted, including limited response for two children. Some behaviors occurred too infrequently at baseline to show clear change, and others were monitored but not specifically targeted. This pattern suggests caregivers distinguished among behavior changes rather than reporting that everything improved, but it does not rule out expectations influencing their reports. Technology problems and reporting demands limited data collection. These findings support tailoring goals and the amount of coaching to each family's needs.
Sustained benefit requires further study. At follow-up, the first group's child-behavior scores remained somewhat lower than at the beginning, but those differences were no longer statistically significant. This finding and caregivers' requests for continued support warrant evaluating booster sessions and ongoing coaching. The study relied largely on caregiver reports and analyzed families who completed all assessments. Its modest sample also limits certainty about smaller effects: nonsignificant differences do not prove that the program and usual services were equivalent. Larger studies may help, but recruiting many more families is difficult in a rare condition. Carefully designed single-case experiments could test program components repeatedly within individual families and identify which strategies help, for whom, and under what conditions.
Overall, PWS Smart Start offers a feasible and highly acceptable model for connecting families with PWS-specific behavioral support without traveling to a specialty clinic. The randomized findings support preliminary benefits in selected areas, while broader before-and-after improvements and individual behavior patterns justify continued research. The next priority is to determine how to sustain gains and adapt support when progress is limited, while complementing the medical, nutritional, educational, and behavioral services children already receive.
Children with PWS may experience emotional outbursts, repetitive behavior, skin picking, difficulty with changes in routine, and food-related behavioral concerns. These challenges can affect learning, everyday activities, and family life. Caregivers need strategies that account for PWS, yet many families have difficulty finding professionals with relevant expertise. Among caregivers who completed this study, 86% reported that they had never received caregiver training before joining the program. All agreed at the beginning of the study that families need behavioral support tailored to PWS.
PWS Smart Start was delivered through 16 weekly, individual online sessions with a Board-Certified Behavior Analyst. Families participated from their homes in the United States and internationally. Caregivers learned how to understand and track behavior, prevent predictable difficulties, teach communication and other useful skills, and reinforce appropriate behavior. The program also addressed schedules, waiting, self-calming, food security, and ways to use skills across daily routines. Caregivers practiced the strategies, completed activities between sessions, and worked with their coach to develop a plan tailored to their child's strengths and needs.
Sixty-five caregiver-child pairs were randomly assigned to receive the program immediately or to continue their usual services while waiting. Fifty-eight pairs completed all study assessments and were included in the analyses. Children were 3-12 years old. The second group then received PWS Smart Start, allowing researchers to examine changes following participation in both groups. The first group also completed a later assessment to examine whether changes persisted after coaching ended.
Researchers compared each family's reports before and after its training period. These analyses showed a wider range of encouraging changes. Caregivers reported reductions in food-related behavior, repetitive behavior, skin picking, rigidity, disruptive behavior, and the overall interference associated with behavioral symptoms. They also reported less distress in several areas of parenting, lower burnout, greater use of positive and behavior-based parenting strategies, and a modest improvement in family quality of life. Total parenting stress did not change significantly. These before-and-after changes do not establish that the program alone caused the improvements. In the randomized comparison, differences in stress, burnout, family quality of life, and several child-behavior outcomes did not reach statistical significance.
The study also examined whether the program could be delivered consistently online. Coaches completed an average of 99.7% of the required program components. Caregiver-acquisition checklists, which assessed completion of training components and homework, averaged 99.6%. Independent reviewers checked more than one-third of sessions and showed high agreement with the original ratings. These results support the feasibility of providing structured, individualized caregiver training through telehealth. They do not establish that every strategy was used independently or consistently in all daily situations, which remains an important question for future research.
Caregiver feedback was strongly positive. Forty-eight caregivers completed the satisfaction survey. All respondents agreed that the content was relevant, important, and tailored to PWS, and all would recommend the program to other families. About 96% thought the number of sessions was appropriate. Among those answering the relevant questions, all reported learning new skills and feeling better able to help their child. Most reported changing how they arranged the home environment. About 94% wanted continued access to their coach after the program ended, indicating that families valued the support and wanted help applying it over time.
Children's responses varied. Eight caregivers completed frequent reports of individualized behaviors. Four of five children with emotional-dysregulation targets improved after caregivers completed the emotional-regulation module, although behavior remained variable and the study did not independently test that module. Other concerns persisted, including limited response for two children. Some behaviors occurred too infrequently at baseline to show clear change, and others were monitored but not specifically targeted. This pattern suggests caregivers distinguished among behavior changes rather than reporting that everything improved, but it does not rule out expectations influencing their reports. Technology problems and reporting demands limited data collection. These findings support tailoring goals and the amount of coaching to each family's needs.
Sustained benefit requires further study. At follow-up, the first group's child-behavior scores remained somewhat lower than at the beginning, but those differences were no longer statistically significant. This finding and caregivers' requests for continued support warrant evaluating booster sessions and ongoing coaching. The study relied largely on caregiver reports and analyzed families who completed all assessments. Its modest sample also limits certainty about smaller effects: nonsignificant differences do not prove that the program and usual services were equivalent. Larger studies may help, but recruiting many more families is difficult in a rare condition. Carefully designed single-case experiments could test program components repeatedly within individual families and identify which strategies help, for whom, and under what conditions.
Overall, PWS Smart Start offers a feasible and highly acceptable model for connecting families with PWS-specific behavioral support without traveling to a specialty clinic. The randomized findings support preliminary benefits in selected areas, while broader before-and-after improvements and individual behavior patterns justify continued research. The next priority is to determine how to sustain gains and adapt support when progress is limited, while complementing the medical, nutritional, educational, and behavioral services children already receive.
Funded Year:
2022
Awarded to:
Kasey Bedard, Ph.D.
Amount:
$152,667
Institution:
The Chicago School of Professional Psychology
Researcher:
