ArticleThe European journal of health economics : HEPAC : health economics in prevention and care2025
Health state utilities associated with X-linked retinitis pigmentosa (XLRP).
Article in The European journal of health economics : HEPAC : health economics in prevention and care, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
3 citing papers in PubMed.
- The quality-of-life impacts and economic burden of X-linked retinitis pigmentosa caused by variants in RPGR.Eye (London, England) · 2026Review
- X-Linked Retinitis Pigmentosa (XLRP): An Investigation of the Impact of XLRP on Health-Related Quality of Life of Carers in Great Britain.Advances in therapy · 2026Article
- Content Validity of Two Generic Preference-Based Measures in Retinitis Pigmentosa, Including X-Linked Retinitis Pigmentosa.PharmacoEconomics - open · 2026Article
Corrections and comments
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Authors and funding
10 authors.
Funding
Abstract
backgroundX-linked retinitis pigmentosa (XLRP) is a rare, inherited retinal disease characterized by impairment in visual field and visual acuity with continuous progression leading to blindness. Gene therapies for XLRP are under investigation, and health state utilities are needed for use in cost-utility analyses examining the value of these treatments.
objectiveThis study aimed to estimate utilities associated with XLRP severity.
methodsEleven health state vignettes depicting combinations of impairment in visual field and visual acuity associated with XLRP were developed based on literature review and input from clinicians, patients, and a caregiver. Vignettes included text describing visual acuity impairment, visual field impairment, night blindness, impact on quality of life, and two images representing the combination of visual field and visual acuity impairment for each health state. Health states were valued in time trade-off interviews with general population respondents in the UK.
resultsA total of 245 participants completed interviews (51.0% female; mean age = 41.4 years; Newcastle, n = 80; London, n = 85; Edinburgh, n = 80). In a ranking task, participants preferred health states with less severe visual impairment, and this preference was reflected in the utilities. Mean (standard deviation) utilities ranged from 0.900 (0.121) for the health state with no visual acuity impairment and mild visual field impairment to 0.271 (0.478) for the health state describing blindness.
conclusionResults highlight the substantial impact of visual impairment on health state preference and quality of life. The health state utilities estimated in this study may be appropriate for use in cost-effectiveness models evaluating treatments for XLRP. JEL CLASSIFICATION CODES: I1; I12; I19.
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