Powell, P. orcid.org/0000-0003-1169-3431, Carlton, J., Peasgood, T. et al. (3 more authors) (2025) Understanding the health-related quality of life impacts of caring for children and adolescents with rare progressive life-limiting conditions: key challenges and future research priorities. Quality of Life Research. ISSN 0962-9343
Abstract
The inclusion of health-related quality of life (HRQoL) impacts on informal carers in health technology assessments (HTAs) is lacking due, primarily, to a deficiency in evidence and methodological issues on how informal carer HRQoL is captured and incorporated into economic models. These issues are magnified in areas of significant burden, such as caring for children and adolescents with rare, progressive, life-limiting conditions. In this commentary we outline key challenges in measuring, and incorporating in HTA submissions, informal carer HRQoL data in rare, progressive, paediatric, life-limiting conditions and identify future research priorities in this space. We argue that: (i) a generic model of carer HRQoL is likely inadequate to characterise the HRQoL impacts in this population; (ii) the underlying evidence-base is underdeveloped, including understanding commonalities across conditions, impact beyond the primary carer, and differences over time; and (iii) methodological challenges in modelling informal carer HRQoL in cost-effectiveness analysis are inhibiting progress. A research agenda is proposed that addresses these challenges by focusing first on in-depth qualitative research to develop an appropriate, content valid ‘disease-group-specific’ model for understanding informal carer HRQoL in rare, progressive, paediatric, life-limiting conditions. This model can be used to inform the appropriate measurement of carer HRQoL in this population, which, alongside methodological research on addressing modelling challenges, can help to facilitate the recommended inclusion of informal carer HRQoL data in HTA submissions for children and adolescents living with rare, progressive, life-limiting conditions.
Metadata
Item Type: | Article |
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Authors/Creators: |
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Copyright, Publisher and Additional Information: | © The Author(s) 2025. 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: | Carers; Children; Rare disease; Health-related quality of life; Health technology assessment |
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 |
Depositing User: | Symplectic Sheffield |
Date Deposited: | 05 Mar 2025 16:20 |
Last Modified: | 05 Mar 2025 16:20 |
Status: | Published online |
Publisher: | Springer |
Refereed: | Yes |
Identification Number: | 10.1007/s11136-025-03937-8 |
Open Archives Initiative ID (OAI ID): | oai:eprints.whiterose.ac.uk:224035 |