Exploring Communication Ability in Individuals With Angelman Syndrome: Findings From Qualitative Interviews With Caregivers.


Communication deficits have a substantial impact on quality of life for individuals with Angelman syndrome (AS) and their families, but limited qualitative work exists to support the necessary content of measures aiming to assess communication for these individuals. Following best practices for concept elicitation studies, we conducted individual qualitative interviews with caregivers and clinicians to elicit meaningful aspects of communication for individuals with AS. Caregivers were able to discuss their child's specific communication behaviors within a large number of expressive, receptive, and pragmatic functions via numerous symbolic and non-symbolic modalities. These results aligned well with published literature on communication in AS and will be used to inform the design of a novel caregiver-reported measure. Future studies on communication in individuals with AS should focus on gathering quantitative data from large samples of diverse caregivers, which would allow for estimations of the frequency of specific behaviors across the population.





Published Version (Please cite this version)


Publication Info

Zigler, Christina K, Nicole Lucas, Molly McFatrich, Kelly L Gordon, Harrison N Jones, Allyson Berent, Jennifer Panagoulias, Paula Evans, et al. (2023). Exploring Communication Ability in Individuals With Angelman Syndrome: Findings From Qualitative Interviews With Caregivers. American journal on intellectual and developmental disabilities, 128(3). pp. 185–203. 10.1352/1944-7558-128.3.185 Retrieved from https://hdl.handle.net/10161/27289.

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Christina Kelsey Zigler

Adjunct Associate Professor in Population Health Sciences

Dr. Zigler is an independent investigator dedicated to improving the ways we measure patient-centered, meaningful health states, specializing in pediatrics, rare diseases, and clinical trials. She also is a methodological co-investigator who supports the application of novel statistical approaches and measurement strategies across a diverse array of studies.

Her methodological expertise includes established qualitative and quantitative techniques (i.e. concept elicitation, cognitive testing, classical test theory, item-response theory) alongside innovative methods (i.e. novel ways to identify meaningful change), to ensure the measures are ultimately centered in the patients’ meaningful experiences. Further, she works in a variety of populations and contexts, including pediatrics, where patient-reported outcome measures empower children to report directly on their own health status, and rare diseases, where a creative and pragmatic approach to measure development is necessary and patient-centeredness is essential.

Areas of Expertise: Health Measurement, Health Behavior, and Health Services Research

Harrison N. Jones

Associate Professor of Head and Neck Surgery & Communication Sciences

Bryce B. Reeve

Professor in Population Health Sciences

Dr. Bryce Reeve is a Professor of Population Health Sciences and Professor of Pediatrics at Duke University School of Medicine.  He also serves as Director of the Center for Health Measurement since 2017.  Trained in psychometric methods, Dr. Reeve’s work focuses on assessing the impact of disease and treatments on the lives of patients and their caregivers.  This includes the development of clinical outcome assessments using both qualitative and quantitative methods, and the integration of patient-centered data in research and healthcare delivery settings to inform decision-making.  From 2000 to 2010, Dr. Reeve served as Program Director for the U.S. National Cancer Institute and oversaw a portfolio of health-related quality of life research in cancer patients. From 2010 to 2017, he served as Professor of Health Policy and Management at the University of North Carolina.  From 2011-2013, Dr. Reeve served as President of the International Society for Quality of Life Research (ISOQOL).  In 2015, he received the John Ware and Alvin Tarlov Career Achievement Prize in Patient-Reported Outcomes Measures.  In 2017, 2018, 2019 and 2021, he was ranked in the top 1% most-cited in his respective field over the past 11-year period.

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