Purpose: Huntington disease (HD) is a chronic, debilitating genetic disease that affects physical, emotional, cognitive, and social health. Existing patient-reported outcomes (PROs) of health-related quality of life (HRQOL) used in HD are neither comprehensive, nor do they adequately account for clinically meaningful changes in function. While new PROs examining HRQOL (i.e., Neuro-QoL-Quality of Life in Neurological Disorders and PROMIS-Patient-Reported Outcomes Measurement Information System) offer solutions to many of these shortcomings, they do not include HD-specific content, nor have they been validated in HD. HDQLIFE addresses this by validating 12 PROMIS/Neuro-QoL domains in individuals with HD and by using established PROMIS methodology to develop new, HD-specific content.
Methods: New item pools were developed using cognitive debriefing with individuals with HD, and expert, literacy, and translatability reviews. Existing item banks and new item pools were field tested in 536 individuals with prodromal, early-, or late-stage HD.
Results: Moderate to strong relationships between Neuro-QoL/PROMIS measures and generic self-report measures of HRQOL, and moderate relationships between Neuro-QoL/PROMIS and clinician-rated measures of similar constructs supported the validity of Neuro-QoL/PROMIS in individuals with HD. Exploratory and confirmatory factor analysis, item response theory, and differential item functioning analyses were utilized to develop new item banks for Chorea, Speech Difficulties, Swallowing Difficulties, and Concern with Death and Dying, with corresponding six-item short forms. A four-item short form was developed for Meaning and Purpose.
Conclusions: HDQLIFE encompasses both validated Neuro-QoL/PROMIS measures, as well as five new scales in order to provide a comprehensive assessment of HRQOL in HD.
Download full-text PDF |
Source |
|---|---|
| http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6108175 | PMC |
| http://dx.doi.org/10.1007/s11136-016-1386-3 | DOI Listing |
Publication Analysis
Top Keywords
Similar Publications
Under-five mortality and social determinants in africa: a systematic review.
Eur J Pediatr
January 2025
Global Health and Tropical Medicine, GHTM, LA-REAL, Instituto de Higiene e Medicina Tropical, IHMT, Universidade NOVA de Lisboa, Lisbon, Portugal.
Purpose: Under-five mortality is a key public health indicator, highly responsive to preventive interventions. While global efforts have made strides in reducing mortality rates in this age group, significant disparities persist, particularly in Sub-Saharan Africa. This study aimed to systematically review the factors influencing under-five mortality in Africa, focusing on sociodemographic factors and health-related determinants.
View Article and Find Full Text PDFUnleashing creativity in people with Parkinson's disease: a pilot study of a co-designed creative arts therapy.
J Neurol
January 2025
Vienna Cognitive Science Hub, University of Vienna, Vienna, Austria.
Background: Conventional medical management, while essential, cannot address all multifaceted consequences of Parkinson's disease (PD). This pilot study explores the potential of a co-designed creative arts therapy on health-related quality of life, well-being, and pertinent non-motor symptoms.
Methods: We conducted an exploratory pilot study with a pre-post design using validated questionnaires.
Effects of chronic diseases on health related quality of life is mediated by sleep difficulty in middle aged and older adults.
Sci Rep
January 2025
Department of Chronic and Non-Communicable Disease, Hangzhou Center for Disease Control and Prevention (Hangzhou Health Supervision Institution), Hangzhou, Zhejiang Provinces, People's Republic of China.
Middle-aged and older adults with chronic diseases are more likely to encounter sleep difficulty and have a reduced Health-Related Quality of Life (HRQoL), but there is little research on their possible mechanisms. Therefore, the main objective of this study was to explore how sleep difficulty mediates the impact of chronic diseases on the HRQoL of middle-aged and older adults. The survey data were from a cross-sectional study carried out in 2019 in Hangzhou, China.
View Article and Find Full Text PDFQuality of life and return to work rates in the first two years following major burn injury.
Burns
December 2024
School of Public Health and Preventive Medicine, Monash University, Australia; Population Data Science, Swansea University, Wales.
Background: A better understanding of how major burns patients recover following injury is vital in assessing trauma care and informing healthcare and rehabilitation provision. We aimed to describe the longer-term health and return to work status of major burns patients and identify factors associated with positive outcomes i.e.
View Article and Find Full Text PDFTrajectories of health-related quality of life across age cohorts: A longitudinal analysis of the German population over 16 years.
Soc Sci Med
January 2025
Department Health and Prevention, Institute of Psychology, University of Greifswald, Germany; German Center for Child and Adolescent Health (DZKJ), partner site Greifswald/Rostock, Greifswald, Germany.
Background: Understanding the trajectories of health-related quality of life (HRQoL) across different age cohorts and sociodemographic factors is crucial for promoting healthy aging. This study aims to examine the course of physical and mental HRQoL over a 16-year period in a nationwide sample of the German population.
Methods: Data from the German Socio-Economic Panel spanning from 2002 to 2018 (N = 4111; female = 52.
Want AI Summaries of new PubMed Abstracts delivered to your In-box?
Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!