Evidence map›Paper›PMID 36948373›Full record

ArticleAmerican journal of ophthalmology2023

Change in Cone Structure Over 24 Months in USH2A-Related Retinal Degeneration.

Jacque L Duncan, Wendi Liang, Maureen G Maguire, Travis C Porco, Jessica Wong, Isabelle Audo, Jenna A Cava, Kate Grieve, Angelos Kalitzeos, Joseph Kreis and 5 more

Open access · hybridAbstract readMulticenter Study
In one paragraph

Article in American journal of ophthalmology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.

0numbers the graph read from it
0cells of the map it votes in
11citing papers in PubMed
3.6field-weighted citation impact, top 7% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

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.

2 · The registry

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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

11 citing papers in PubMed, 13 citations in OpenAlex.

  1. Trial
  2. Article
  3. Article
  4. Article
  5. Article
  6. Long-term natural history of ellipsoid zone width inThe British journal of ophthalmology · 2025
    Article
  7. Review
  8. Article
  9. Modeling Human Macular Cone Photoreceptor Spatial Distribution.Investigative ophthalmology & visual science · 2024
    Article
  10. Review
  11. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

15 authors at 5 institutions in 3 countries.

Jacque L DuncanUniversity of California, San Francisco, CA, USA. Electronic address: jacque.duncan@ucsf.edu.
Wendi LiangJaeb Center for Health Research, Tampa, FL, USA.
Maureen G MaguireJaeb Center for Health Research, Tampa, FL, USA.
Travis C PorcoUniversity of California, San Francisco, CA, USA.
Jessica WongUniversity of California, San Francisco, CA, USA.
Isabelle AudoQuinze Vingts National Ophthalmology Hospital, Paris, France.
Jenna A CavaMedical College of Wisconsin, Milwaukee, WI, USA.
Kate GrieveQuinze Vingts National Ophthalmology Hospital, Paris, France.
Angelos KalitzeosMoorfields Eye Hospital, London, UK.
Joseph KreisMedical College of Wisconsin, Milwaukee, WI, USA.
Michel MichaelidesMoorfields Eye Hospital, London, UK.
Nathaniel NorbergQuinze Vingts National Ophthalmology Hospital, Paris, France.
Michel PaquesQuinze Vingts National Ophthalmology Hospital, Paris, France.
Joseph CarrollMedical College of Wisconsin, Milwaukee, WI, USA.
Foundation Fighting Blindness Consortium Investigator Group
Centre hospitalier national d'ophtalmologie des Quinze-Vingts · FRMedical College of Wisconsin · USUniversity of California, San Francisco · USJaeb Center for Health Research · USMoorfields Eye Hospital · GB

Funding

Rapid-Prototyping and Design CoreP30EY002162 · NEI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI ULLIAN, ERIK M · 1985 to 2024
$16.8M
Advanced Technology to Study Visual Function on a Cellular ScaleR01EY023591 · NEI · UNIVERSITY OF CALIFORNIA BERKELEY · PI DUNCAN, JACQUE LYNNE, ROORDA, AUSTIN · 2014 to 2023
$11.2M
Developing Cone-Dominant Retinal Disease Models as a Resource for Translational Vision ResearchU24EY029891 · NEI · MEDICAL COLLEGE OF WISCONSIN · PI CARROLL, JOSEPH, DUNCAN, JACQUE LYNNE · 2018 to 2022
$6.6M
Foundation Fighting Blindness RUSH2A - CONSORTIUM - 2016NEI NIH HHS P30 EY002162NEI NIH HHS R01 EY023591NEI NIH HHS U24 EY029891
6 · The paper itself

Abstract

purposeTo describe cone structure changes using adaptive optics scanning laser ophthalmoscopy (AOSLO) in the Rate of Progression of USH2A-related Retinal Degeneration (RUSH2A) study.

designMulticenter, longitudinal natural history study.

methodsAOSLO images were acquired at 4 centers, twice at baseline and annually for 24 months in this natural history study. For each eye, at least 10 regions of interest (ROIs) with ≥50 contiguous cones were analyzed by masked, independent graders. Cone spacing Z-scores, standard deviations from the normal mean at the measured location, were compared between graders and tests at baseline. The association of cone spacing with clinical characteristics was assessed using linear mixed effects regression models weighted by image quality score. Annual rates of change were calculated based on differences between visits.

resultsFourteen eyes of 14 participants were imaged, with 192 ROIs selected at baseline. There was variability among graders, which was greater in images with lower image quality score (P < .001). Cone spacing was significantly correlated with eccentricity, quality score, and disease duration (P < .02). On average, the cone spacing Z-score increased 0.14 annually (about 9%, P < .001). We observed no significant differences in rate of change between disease type (Usher syndrome or retinitis pigmentosa), imaging site, or grader.

conclusionsUsing current methods, the analysis of quantitative measures of cone structure showed some challenges, yet showed promise that AOSLO images can be used to characterize progressive change over 24 months. Additional multicenter studies using AOSLO are needed to advance cone mosaic metrics as sensitive outcome measures for clinical trials. NOTE: Publication of this article is sponsored by the American Ophthalmological Society.

Indexed as

Retinal DegenerationUsher SyndromesExtracellular Matrix ProteinsHumansOphthalmoscopyRetinal Cone Photoreceptor CellsTomography, Optical CoherenceExtracellular Matrix ProteinsUSH2A protein, human

Identifiers

PMID36948373
PMCPMC11087021
OpenAlexW4328136901

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

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.