Bamber, R. orcid.org/0009-0006-1615-1817, Stavroulakis, T. orcid.org/0000-0002-3535-7822, McDermott, C. orcid.org/0000-0002-1269-9053 et al. (1 more author) (2025) Health-related quality of life of informal carers in ALS: a systematic review of person reported outcome measures. Quality of Life Research. ISSN 0962-9343
Abstract
Purpose
Amyotrophic Lateral Sclerosis (ALS) is a fatal neurodegenerative condition with swift progression. The devastating impact of ALS affects the health-related quality of life (HRQoL) of informal carers. Various person reported outcome measures (PROMs) have been used to assess HRQoL in informal carers in ALS, yet their validity remains unclear. This review aimed to identify and evaluate the content validity of HRQoL PROMs for informal carers in ALS.
Methods
This review was conducted according to best practice COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) methodology. Two literature searches were conducted in November 2023 and April 2024 across MEDLINE, PsycINFO, Embase, CINAHL, the Cochrane Database of Systematic Reviews, CENTRAL and Google Scholar, to identify HRQoL PROMs used with informal carers in ALS, PROM development articles, and psychometric literature. Evidence synthesis followed COSMIN guidance.
Results
12,276 articles were screened, and 109 PROMs were identified, with 43 undergoing full COSMIN assessment. Content validity ratings were ‘Inconsistent’ or ‘Insufficient’ for all PROMs. All PROMs, except the CarerQoL, were rated ‘Insufficient’ for comprehensiveness. Only 18.6% of PROMs included informal carers in development. Quality of evidence supporting content validity ratings was ‘Very Low’ for 93% of PROMs.
Conclusion
HRQoL PROMs used with informal carers in ALS lack evidence to support their content validity, restricting their utility for this purpose. Existing literature on the impact of caring in ALS on informal carers’ HRQoL should be interpreted cautiously. Further research is required to establish the content validity of HRQoL PROMs used for this cohort.
Metadata
Item Type: | Article |
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Authors/Creators: |
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Copyright, Publisher and Additional Information: | © The Author(s) 2025. Open Access: This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. |
Keywords: | Amyotrophic lateral sclerosis; Motor neuron disease; COSMIN; Caring; Carers; Quality of life; Health-related quality of life; Content Validity; Systematic review; Person reported outcome measure |
Dates: |
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Institution: | The University of Sheffield |
Academic Units: | The University of Sheffield > Faculty of Medicine, Dentistry and Health (Sheffield) > School of Medicine and Population Health The University of Sheffield > Faculty of Medicine, Dentistry and Health (Sheffield) > Department of Neuroscience (Sheffield) |
Funding Information: | Funder Grant number DEPARTMENT OF HEALTH AND SOCIAL CARE NIHR301648 |
Depositing User: | Symplectic Sheffield |
Date Deposited: | 02 Jul 2025 10:33 |
Last Modified: | 02 Jul 2025 10:33 |
Status: | Published online |
Publisher: | Springer Science and Business Media LLC |
Refereed: | Yes |
Identification Number: | 10.1007/s11136-025-04012-y |
Open Archives Initiative ID (OAI ID): | oai:eprints.whiterose.ac.uk:228618 |