Abstract
Purpose: In Huntington disease (HD), motor, cognitive, and psychiatric changes can have a detrimental impact on health-related quality of life (HRQOL). The purpose of this paper is to describe the extent and type of assistance needed to complete online HRQOL surveys, and the impact of assistance on HRQOL scores. Methods: A patient-reported outcome measurement system was developed for HD-specific HRQOL. Individuals across the prodromal and diagnosed disease severity spectrum (n = 532) completed surveys by computer, and reported the amount and type of assistance they received. Results: Some participants (n = 56; 10.5%) did not complete all surveys; this group had larger proportions with late stage disease, racial/ethnic minority status, low education and single marital status, and poorer motor, independence and cognitive function compared to those who completed all surveys (n = 476). Overall, 72% of individuals did not receive assistance, 11% received computer assistance only, and 17% received assistance answering the survey questions. The majority of late stage individuals (78%) received some assistance compared to early stage (29%) and prodromal individuals (OpenSPiltSPi 1%). Those who received assistance had higher proportions with late stage disease, were older, had less education, and had poorer functional and cognitive skills. Before and after adjustment for sociodemographic and clinical characteristics, those who received assistance had poorer scores on some HRQOL outcomes than those who did not receive assistance. Conclusions: Computer-based assessments are feasible for many persons with HD, although other methods may also be needed. Clinicians and researchers should develop strategies to assist people with HD to complete HRQOL surveys.
Original language | English (US) |
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Pages (from-to) | 801-810 |
Number of pages | 10 |
Journal | Quality of Life Research |
Volume | 27 |
Issue number | 3 |
DOIs | |
State | Published - Mar 1 2018 |
Funding
Acknowledgements Funding for this project was provided by grant #R01-NS077946 from the National Institutes of Health (NIH) National Institute of Neurological Disorders and Stroke, and the NIH National Center for Advancing Translational Sciences (grant #UL1-TR000433). Part of this study sample was obtained in cooperation with the Predict-HD study. The Predict-HD study was supported by the NIH National Institute of Neurological Disorders and Stroke (grant #R01-NS040068), the NIH-funded Center for Inherited Disease Research (provided support for sample phenotyping), and the CHDI Foundation (award to the University of Iowa). We thank the University of Iowa, the investigators and coordinators of this study, the study participants, the National Research Roster for Huntington Disease Patients and Families, the Huntington Study Group, and the Huntington’s Disease Society of America. We acknowledge the assistance of Jeffrey D. Long, Hans J. Johnson, Jeremy H. Bockholt, and Roland Zschiegner. We also acknowledge Roger Albin, Kelvin Chou and Henry Paulsen for the assistance with participant recruitment. The content is solely the responsibility of the authors and does not necessarily represent the official views of the NIH. HDQLIFE Site Investigators and Coordinators: Noelle Carlozzi, Praveen Dayalu, Stephen Schilling, Amy Austin, Matthew Canter, Siera Goodnight, Jennifer Miner, Nicholas Migliore (University of Michigan, Ann Arbor, MI); Jane Paulsen, Nancy Downing, Isabella DeSoriano, Courtney Shadrick, Amanda Miller (University of Iowa, Iowa City, IA); Kimberly Quaid, Melissa Wesson (Indiana University, Indianapolis, IN); Christopher Ross, Gregory Churchill, Mary Jane Ong (Johns Hopkins University, Baltimore, MD); Susan Perlman, Brian Clemente, Aaron Fisher, Gloria Obialisi, Michael Rosco (University of California Los Angeles, Los Angeles, CA); Michael McCormack, Humberto Marin, Allison Dicke (Rutgers University, Piscataway, NJ); Joel Perlmutter, Stacey Barton, Shineeka Smith (Washington University, St. Louis, MO); Martha Nance, Pat Ede (Struthers Parkinson’s Center); Stephen Rao, Anwar Ahmed, Michael Lengen, Lyla Mourany, Christine Reece (Cleveland Clinic Foundation, Cleveland, OH); Michael Geschwind, Joseph Winer (University of California – San Francisco, San Francisco, CA); David Cella, Richard Gershon, Elizabeth Hahn, Jin-Shei Lai (Northwestern University, Chicago, IL). have received research grants from the National Institutes of Health. Author Stout has received consultancy payments from the University of Michigan. Author Carlozzi has received research grants from the National Institutes of Health and consultancy payments from Teva Pharmaceuticals. Funding Funding for this project was provided by grant #R01-NS077946 from the National Institutes of Health (NIH) National Institute of Neurological Disorders and Stroke, and the NIH National Center for Advancing Translational Sciences (grant #UL1-TR000433).
Keywords
- Health-related quality of life
- Huntington disease
- Patient-reported outcomes
- Surveys and questionnaires
ASJC Scopus subject areas
- Public Health, Environmental and Occupational Health