In their own voices: Developing a self-report measure of Hyperphagia for Individuals with PWS

Funding Summary

Currently, hyperphagia is often assessed by proxy informants on the Hyperphagia Questionnaires. Leveraging insights from previous research -- and with input from a PWS Advisory Board, PWS focus groups and our own experience in developing other PWS-specific measures—this project will develop a self-report measure of hyperphagic symptoms for individuals with PWS (MY-HQ). In doing so, we aim to circumvent their cognitive processing difficulties and tendencies to be secretive about their food-seeking.

Dr. Theresa Strong, Director of Research Programs, shares details on this project in this short video clip. 

Lay Abstract

PWS features a distinctive, complex and hard-to-measure behavioral phenotype, including hyperphagia and related symptoms. Existing measures of hyperphagia have both strengths and limitations. Currently, hyperphagia is often assessed by proxy informants on the Hyperphagia Questionnaires. Although successfully used in clinical trials, the HQ-CT can be vulnerable to informant bias. Until now, researchers have shied away from relying on self-reported hyperphagic symptoms from individuals with PWS. Reasons for doing so are twofold. First, persons with PWS have varying levels of intellectual disabilities (ID) and executive functioning deficits that may deter them from accurate self-reporting (Chevalere et al., 2015). Second, individuals with PWS are prone to sneaking food, manipulating others or bargaining for food, and they want to avoid getting in trouble for these behaviors. As such, they may lie about, distort or deny consuming food not on their diet plans or be secretive about their food-seeking behaviors. Even so, individuals with PWS can still provide unique observations about their hyperphagia. From our interview study (Dykens, Roof, Hunt-Hawkins, 2021), we garnered insights from participants with PWS that rule out one approach to self-reporting their hyperphagic symptoms, while also opening up other promising avenues. Participants were quite informative about how their “hunger urges” or food-seeking impeded their everyday life and future goals. They offered concrete examples of how hunger and food availability interfered with learning or socializing at school, getting along with family and others, and living or working in the community. Leveraging these insights -- and with input from a PWS Advisory Board, PWS focus groups and our own experience in developing other PWS-specific measures—this Project will develop a self-report measure of hyperphagic symptoms for individuals with PWS (MY-HQ). In doing so, we aim to circumvent their cognitive processing difficulties and tendencies to be secretive about their food-seeking. This application is highly innovative in three ways. First, we plan to develop the first ever, psychometrically sound self-report of hyperphagia for persons with PWS, as well as identify barriers that impede responsivity in some individuals. Second, we will involve, for the first time ever, persons with PWS in the research process using Participatory Action Research (PAR). We aim to develop and Assess the Psychometric Properties of MY-HQ. This Aim leverages best-practices in developing questionnaires for persons with ID, the input of a PWS Advisory Board, PWS focus groups, and our expertise in developing other PWS-specific measures (HQ, Food Safe Zone, PWS Profile). In three phases, we will pilot test and evaluate the reliability and validity of MY-HQ via RedCap, an online data collection platform with audio and video enhanced capabilities that will optimize engagement and responsivity of participants. We also aim to uncover reasons (cognitive functioning, executive functioning and other comorbid diagnoses) for non-responders may have difficulty responding to measures of hyperphagia in clinical trials.