ArticleQuality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation2018
Understanding patient-reported outcome measures in Huntington disease: at what point is cognitive impairment related to poor measurement reliability?
Article in Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.
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Who cites it
13 citing papers in PubMed.
- Huntington Disease Health Related Quality of Life, Function and Well Being: The Patient's Perspective.Neurology and therapy · 2025Article
- Outcomes misaligned in mitochondrial encephalomyopathy, lactic acidosis and stroke-like episodes (MELAS): implications for trial design.Brain communications · 2025Article
- Cognitive impairment predicts medication discrepancies in Huntington's Disease: patient self-report compared to pharmacy records.Journal of neurology · 2024Article
- Influence of anosognosia on patient-reported outcomes for psychiatric symptoms and quality of life in Huntington's disease.Parkinsonism & related disorders · 2024Article
- Disease Burden of Huntington's Disease (HD) on People Living with HD and Care Partners in Canada.Journal of Huntington's disease · 2022Article
- Psycholinguistic features, design attributes, and respondent-reported cognition predict response time to patient-reported outcome measure items.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2021Article
- Responsiveness to change over time and test-retest reliability of the PROMIS and Neuro-QoL mental health measures in persons with Huntington disease (HD).Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2020Article
- Management of dysphagia in Huntington's disease: a descriptive review.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2020Review
- Psychometric properties and responsiveness of Neuro-QoL Cognitive Function in persons with Huntington disease (HD).Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2020Article
- HDQLIFE and neuro-QoL physical function measures: Responsiveness in persons with huntington's disease.Movement disorders : official journal of the Movement Disorder Society · 2020Article
- Preliminary development of proxy-rated quality-of-life scales for children and adults with Niemann-Pick type C.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2019Article
- Positive Affect and Well-Being in Huntington's Disease Moderates the Association Between Functional Impairment and HRQOL Outcomes.Journal of Huntington's disease · 2019Article
- Validation of Neuro-QoL and PROMIS Mental Health Patient Reported Outcome Measures in Persons with Huntington Disease.Journal of Huntington's disease · 2019Article
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Authors and funding
6 authors.
Funding
Abstract
purposeSymptom progression in Huntington disease (HD) is associated with cognitive decline which may interfere with the self-report of symptoms. Unfortunately, data to support or refute the psychometric reliability of patient-reported outcomes (PROs) as HD progresses are limited. This is problematic given that PROs are increasingly recognized as important measures of efficacy for new treatments.
methodsWe examined PRO data from the HDQLIFE Measurement System (Speech Difficulties; Swallowing Difficulties; Chorea) in 509 individuals with premanifest, early-stage, or late-stage HD. Clinician-administered assessments of motor functioning (items from the UHDRS) and standardized objective assessments of cognition (Stroop, Symbol Digit Modalities) were also collected. We examined item bias using differential item functioning (DIF) across HD stage (premanifest, early-, late-) and relative to cognitive performance. We also examined the correlations between self-report and clinician ratings. Regression models that considered total cognitive ability were utilized to determine psychometric reliability of the PROs.
resultsMost PRO items were free from DIF for both staging and cognition. There were modest correlations between PROs and clinician report (ranged from - 0.40 to - 0.60). Modeling analyses indicated that psychometric reliability breaks down with poorer cognition and more progressed disease stage; split-half reliability was compromised (i.e., split-half reliability < 0.80) when scores were < 136 for Chorea, < 109 for Speech Difficulties, and < 179 for Swallowing Difficulties.
conclusionsResults indicate that the psychometric reliability of PROs can be compromised as HD symptoms progress and cognition declines. Clinicians should consider PROs in conjunction with other types of assessments when total cognition scores exceed critical thresholds.
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