Foundation Grant Helps Fund Development of Self-reported Survey for PWS Patients

Clinical tests of excessive hunger have typically relied on caregiver feedback

Marisa Wexler, MS avatar

by Marisa Wexler, MS |

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The Foundation for Prader-Willi Research has awarded a two-year, $141,444 grant to help fund the development of a patient-reported questionnaire to assess the impact of hunger for people with Prader-Willi syndrome (PWS).

The project is being led by Elisabeth Dykens, PhD, a professor of psychology and human development at Vanderbilt Peabody College.

This project is a first when it comes to including people with PWS in the research process, according to Dykens. The project is built on Participatory Action Research, or PAR — a strategy that involves facilitating active collaborations between researchers and participants in studies.

“The central principle of PAR is ‘Nothing about us without us.’ Our collaborators with PWS are experts by experience and integral to this project,” Dykens said in a university press release. “By adopting a collaborative approach, we combine the skills of our research team and the perspectives of those with PWS to create a product that neither group could do alone.”

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Researchers aim to create a questionnaire that’s accessible for people with PWS

A hallmark symptom of PWS is hyperphagia, or uncontrolled hunger. When researchers are designing clinical trials to test potential treatments for PWS, it’s critical to have accurate ways to measure how severe participants’ hyperphagia is, and how much this symptom is causing problems in their daily life.

People with PWS often have intellectual differences from their unaffected peers, and they commonly have trouble with executive function — which refers to high-order cognitive abilities such as working memory, cognitive flexibility, planning, and reasoning — and behavior regulation. Plus, hyperphagia often drives them to sneak food, which can lead to feelings of guilt and, consequently, dishonesty or distortion about their food consumption.

For these reasons, clinical assessments of hyperphagia in PWS have generally relied on proxy informants, such as parents or other caregivers, rather than involving the person who is actually experiencing the symptom.

With this new project, the researchers aim to create a questionnaire assessing hyperphagia symptoms that’s specifically designed to be accessible for people with PWS. To design the questionnaire, they will use insights from a 2021 study where Dykens and colleagues conducted interviews with PWS patients to learn more about their experiences. The project will also benefit from insights of a PWS advisory board and focus groups.

The end goal is to make an online tool that’s user-friendly and compatible with a touch screen, and audio- and video-enhanced features to improve utility, which could be used in future studies to help people with PWS have their voices heard when they participate in clinical studies.