A remote play-based intervention program improved pretend play skills, cognition, and behavior in children with Prader-Willi syndrome (PWS), a small study shows.
The preliminary findings support the validation of this remote approach in larger studies of a patient population with a high unmet need for effective behavioral interventions, researchers said.
The study, “Show me what happens next: Preliminary efficacy of a remote play-based intervention for children with Prader-Willi syndrome,” was published in the journal Research in Developmental Disabilities and conducted by scientists at Case Western Reserve University in Cleveland, Ohio.
PWS’s behavioral hallmark is insatiable appetite, but patients often have repetitive behaviors, anger outbursts, intellectual disability, social deficits, and emotional impairment.
All contribute to impaired adaptive functioning, or a harder adaptation to new or difficult circumstances.
Previous research also suggests that children with PWS have impaired pretend play abilities, with low levels of imagination, disorganized storylines, and low emotion expression, similar to children with autism spectrum disorder.
However, the addition of a play partner to these children with PWS led to significant improvements across these domains, suggesting that interventions promoting pretend play may “increase socioemotional understanding and cognitive flexibility while decreasing rigidity and repetitive behaviors,” the researchers wrote.
While studies evaluating potential interventions for PWS patients are greatly needed, the disease’s rarity challenges the recruitment and monitoring of sufficient participants from a local area.
Telehealth, or telemedicine, is the use of technology to connect directly with a healthcare professional from just about anywhere a connection is available.
This approach is gaining importance both in research and clinical practice for the assessment, parent training, and direct intervention for pediatric and adult patient populations. As such, telehealth may reduce the barriers to developing interventions for people with PWS.
In 2017, researchers at Case Western showed that a play-based telehealth intervention was possible in children with PWS, ages 6–12 years, and that they enjoyed the program and looked forward to the sessions. In addition, parents reported high acceptability and considered it a viable option to address the socioemotional problems seen in their children.
The same team now evaluated the effects of such remote play-based intervention in pretend play ability, cognitive flexibility, and behavior in the same 15 PWS patients (eight girls and seven boys).
Participants, with a mean age of 8.8 years, were recruited from locations that were at least two hours away from the main study site to test the feasibility of remote intervention.
In a first in-person visit, children underwent cognitive and behavioral assessments, while the parent completed demographic information and a survey on their child’s overall behavioral functioning.
Children’s cognitive and emotional processes involved in play were assessed through the Affect in Play Scale, a validated measure of pretend play skills, such as the organization of the storyline, imagination and comfort while playing, as well as emotion expression frequency and emotion variety. Creativity and idea generation, or cognitive flexibility, were assessed with the Multidimensional Stimulus Fluency Measure, which uses visual and tactile stimuli and requires verbal responses to questions such as “name as many red things as possible,” “what does an abstract shape look like,” and “how can you use a box.”
The children were given a standardized set of toys and parents a webcam and intervention manual before the remote intervention. The program comprised sessions of 15−20-minutes, in which a researcher played one-on-one with the child over a video-conferencing platform, twice a week for six weeks.
It was divided into three sections of four sessions each, focused on building play skills, emotional expression and understanding, and self-coping behavioral techniques. At the beginning of each section, the researcher and parent had a remote meeting to review the section’s goals and concepts, and parents were encouraged to practice play, socioemotional, and/or behavioral skills with their child at home.
Within four weeks of program completion, both the children and parents completed the same assessments as in the first visit.
Results showed that children with PWS had deficits in pretend play and cognitive flexibility in their first assessment, compared with typically developing children.
“These deficits were characterized by rigid thought patterns (i.e. getting stuck on a topic) and repetitive behavior (i.e. replaying the same storylines),” the researchers wrote.
Notably, cognitive and emotional abilities in pretend play, including imagination, emotion variety, and thematic frequency, improved significantly after the intervention. Organization also showed a trend toward improvement. Imagination, organization, and emotion expression frequency all improved into the normative range seen in unaffected children.
In addition, creativity and idea generation increased significantly post-intervention.
“This study is the first to show preliminary evidence that remote intervention delivered directly to the child may be effective at increasing pretend play ability and enhancing cognitive flexibility in children with PWS,” the researchers wrote.
Given that these skills are associated with socioemotional and social-cognitive functioning, these findings point out “an important new avenue of feasible and accessible behavioral intervention that should be further explored for school-age children with PWS,” the team added.
Larger studies including a control group of children not undergoing the remote intervention are needed to confirm the benefits of this type of approach in PWS patients, the researchers said.
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