SynthesisThe British journal of ophthalmology2021
Long-term natural history of visual acuity in eyes with choroideremia: a systematic review and meta-analysis of data from 1004 individual eyes.
Synthesis in The British journal of ophthalmology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers, 1 of them a synthesis that pooled it.
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.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
20 citing papers in PubMed, 1 synthesis or guideline pooled it, 28 citations in OpenAlex.
- Bilateral visual acuity decline in males with choroideremia: a pooled, cross-sectional meta-analysis.BMC ophthalmology · 2022Pooled it
- Subretinal timrepigene emparvovec in adult men with choroideremia: a randomized phase 3 trial.Nature medicine · 2023Trial
- A 48-Month, Prospective, Multicenter, Natural History Study of Choroideremia.Ophthalmology science · 2026Article
- Validation of Ultra-Widefield Autofluorescence Imaging as an Endpoint for Disease Progression in Choroideremia: A Comparative Study.Ophthalmology science · 2026Article
- Longitudinal and cross-sectional study of retinal phenotypes and visual function in choroideremia carriers: a new grading system.Eye and vision (London, England) · 2026Article
- One down but many more to go: the state of gene therapy for inherited retinal disease.Regenerative medicine · 2025Review
- Longitudinal Assessment of Cone Structure, Choriocapillaris, and Retinal Sensitivity in Choroideremia.Investigative ophthalmology & visual science · 2025Article
- Long-term natural history of ellipsoid zone width inThe British journal of ophthalmology · 2025Article
- Article
- Exploring Scotopic Microperimetry as an Outcome Measure in Choroideremia.Translational vision science & technology · 2024Article
- Parafoveal cone function in choroideremia assessed with adaptive optics optoretinography.Scientific reports · 2024Article
- Using Goldmann Visual Field Volume to Track Disease Progression in Choroideremia.Ophthalmology science · 2023Article
- Choroideremia: The Endpoint Endgame.International journal of molecular sciences · 2023Review
- Cone Photoreceptor Morphology in Choroideremia Assessed Using Non-Confocal Split-Detection Adaptive Optics Scanning Light Ophthalmoscopy.Investigative ophthalmology & visual science · 2023Article
- Natural history of central sparing in geographic atrophy secondary to non-exudative age-related macular degeneration.The British journal of ophthalmology · 2022Article
- A hierarchical Bayesian entry time realignment method to study the long-term natural history of diseases.Scientific reports · 2022Article
- Gene therapy for inherited retinal diseases.Annals of translational medicine · 2021Review
- Autosomal Dominant Gyrate Atrophy-Like Choroidal Dystrophy Revisited: 45 Years Follow-Up and Association with a NovelInternational journal of molecular sciences · 2021Article
- Update on Gene Therapy Clinical Trials for Choroideremia and Potential Experimental Therapies.Medicina (Kaunas, Lithuania) · 2021Review
- Longitudinal Study to Assess the Quantitative Use of Fundus Autofluorescence for Monitoring Disease Progression in Choroideremia.Journal of clinical medicine · 2021Article
Corrections and comments
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Authors and funding
6 authors at 1 institution in 1 country.
Funding
Abstract
BACKGROUND/
aimsBest-corrected visual acuity (BCVA) is the most common primary endpoint in treatment trials for choroideremia (CHM) but the long-term natural history of BCVA is unclear.
methodsWe searched in seven databases to identify studies that reported BCVA of untreated eyes with CHM. We sought individual-level data and performed segmented regression between BCVA and age. For eyes followed longitudinally, we introduced a horizontal translation factor to each dataset to account for different ages at onset of a rapid BCVA decline.
resultsWe included 1004 eyes from 23 studies. BCVA of the right and left eyes was moderately correlated (r=0.60). BCVA as a function of age followed a 2-phase decline (slow followed by rapid decline), with an estimated transition age of 39.1 years (95% CI 33.5 to 44.7). After the introduction of horizontal translation factors to longitudinal datasets, BCVA followed a 2-phase decline until it reached 0 letters (r
conclusionBCVA in eyes with CHM follows a 2-phase linear decline with a transition age of approximately 39 years. Future trials enrolling young patients may not be able to use BCVA as a primary or sole endpoint, but rather, may need to employ additional disease biomarkers that change before age 39. BCVA may still have utility as a primary endpoint for patients older than 39 years who have measurable BCVA decline rates.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.