Journal article
Measuring quality of life in muscular dystrophy
Neurology, Vol.84(10), pp.1034-1042
03/10/2015
DOI: 10.1212/WNL.0000000000001336
PMCID: PMC4352095
PMID: 25663223
Abstract
Objectives: The objectives of this study were to develop a conceptual model of quality of life (QOL) in muscular dystrophies (MDs) and review existing QOL measures for use in the MD population.
Methods: Our model for QOL among individuals with MD was developed based on a modified Delphi process, literature review, and input from patients and patient advocacy organizations. Scales that have been used to measure QOL among patients with MD were identified through a literature review and evaluated using the COSMIN (Consensus-Based Standards for the Selection of Health Measurement Instruments) checklist.
Results: The Comprehensive Model of QOL in MD (CMQM) captures 3 broad domains of QOL (physical, psychological, and social), includes factors influencing self-reported QOL (disease-related factors, support/resources, and expectations/aspirations), and places these concepts within the context of the life course. The literature review identified 15 QOL scales (9 adult and 6 pediatric) that have been applied to patients with MD. Very few studies reported reliability data, and none included data on responsiveness of the measures to change in disease progression, a necessary psychometric property for measures included in treatment and intervention studies. No scales captured all QOL domains identified in the CMQM model.
Conclusions: Additional scale development research is needed to enhance assessment of QOL for individuals with MD. Item banking and computerized adaptive assessment would be particularly beneficial by allowing the scale to be tailored to each individual, thereby minimizing respondent burden.
Details
- Title: Subtitle
- Measuring quality of life in muscular dystrophy
- Creators
- Carla M. Bann - RTI InternationalRichard T. Abresch - Muscular Dystrophy AssociationBarbara Biesecker - Muscular Dystrophy AssociationKristin Caspers Conway - Muscular Dystrophy AssociationChad Heatwole - Muscular Dystrophy AssociationHolly Peay - Muscular Dystrophy AssociationPeter Scal - Muscular Dystrophy AssociationJonathan Strober - Muscular Dystrophy AssociationKaren Uzark - Muscular Dystrophy AssociationJodi Wolff - Muscular Dystrophy AssociationMarjorie Margolis - Muscular Dystrophy AssociationAngela Blackwell - Muscular Dystrophy AssociationNatalie Street - Muscular Dystrophy AssociationAngela Montesanti - Muscular Dystrophy AssociationJulie Bolen - Muscular Dystrophy Association
- Resource Type
- Journal article
- Publication Details
- Neurology, Vol.84(10), pp.1034-1042
- DOI
- 10.1212/WNL.0000000000001336
- PMID
- 25663223
- PMCID
- PMC4352095
- NLM abbreviation
- Neurology
- ISSN
- 0028-3878
- eISSN
- 1526-632X
- Publisher
- Lippincott Williams & Wilkins
- Number of pages
- 9
- Grant note
- ZIAHG200317 / NATIONAL HUMAN GENOME RESEARCH INSTITUTE; United States Department of Health & Human Services; National Institutes of Health (NIH) - USA; NIH National Human Genome Research Institute (NHGRI) T32NS007338 / NATIONAL INSTITUTE OF NEUROLOGICAL DISORDERS AND STROKE; United States Department of Health & Human Services; National Institutes of Health (NIH) - USA; NIH National Institute of Neurological Disorders & Stroke (NINDS) 200-2007-22644/0016 / Centers for Disease Control and Prevention; United States Department of Health & Human Services; Centers for Disease Control & Prevention - USA
- Language
- English
- Date published
- 03/10/2015
- Academic Unit
- Epidemiology
- Record Identifier
- 9984627300902771
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