Evidence map›Paper›PMID 36646238›Full record

ArticleAmerican journal of ophthalmology2023

Cone Structure and Function in RPGR- and USH2A-Associated Retinal Degeneration.

Paul S Micevych, Jessica Wong, Hao Zhou, Ruikang K Wang, Travis C Porco, Joseph Carroll, Austin Roorda, Jacque L Duncan

Open access · hybridAbstract readComparative StudyMulticenter 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 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
1.1field-weighted citation impact, top 24% 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

4 citing papers in PubMed, 7 citations in OpenAlex.

  1. Observational
  2. Article
  3. The Role of Visual Electrophysiology in Systemic Hereditary Syndromes.International journal of molecular sciences · 2025
    Review
  4. 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

8 authors at 4 institutions in 1 country.

Paul S MicevychFrom the Department of Ophthalmology, University of California (P.S.M., J.W., T.C.P., J.L.D.), San Francisco, California.
Jessica WongFrom the Department of Ophthalmology, University of California (P.S.M., J.W., T.C.P., J.L.D.), San Francisco, California.
Hao ZhouDepartment of Bioengineering, University of Washington (H.Z., R.K.W.), Seattle, Washington.
Ruikang K WangDepartment of Bioengineering, University of Washington (H.Z., R.K.W.), Seattle, Washington.
Travis C PorcoFrom the Department of Ophthalmology, University of California (P.S.M., J.W., T.C.P., J.L.D.), San Francisco, California; Francis I. Proctor Foundation, Department of Ophthalmology, University of California (T.C.P.), San Francisco, California.
Joseph CarrollDepartment of Ophthalmology & Visual Sciences, Medical College of Wisconsin Eye Institute (J.C.), Milwaukee, Wisconsin.
Austin RoordaHerbert Wertheim School of Optometry & Vision Science, University of California Berkeley (A.R.), Berkeley, California, USA.
Jacque L DuncanFrom the Department of Ophthalmology, University of California (P.S.M., J.W., T.C.P., J.L.D.), San Francisco, California. Electronic address: jacque.duncan@ucsf.edu.
University of California, San Francisco · USUniversity of Washington · USMedical College of Wisconsin · USUniversity of California, Berkeley · US

Funding

Rapid-Prototyping and Design CoreP30EY002162 · NEI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI LAMBA, DEEPAK ASHOK · 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
Assessing Photoreceptor Structure and Function in Normal and Diseased RetinaeR01EY017607 · NEI · MEDICAL COLLEGE OF WISCONSIN · PI Joseph Carroll · 2008 to 2026
$8.3M
NEI NIH HHS P30 EY002162NEI NIH HHS R01 EY017607NEI NIH HHS R01 EY023591
6 · The paper itself

Abstract

purposeTo compare cone structure and function between RPGR- and USH2A-associated retinal degeneration.

designRetrospective, observational, cross-sectional study.

methodsThis multicenter study included 13 eyes (9 participants) with RPGR-related X-linked retinitis pigmentosa (RPGR), 15 eyes (10 participants) with USH2A-related Usher syndrome type 2 (USH2), 16 eyes (9 participants) with USH2A-related autosomal recessive retinitis pigmentosa (ARRP), and 7 normal eyes (6 participants). Structural measures included cone spacing and density from adaptive optics scanning laser ophthalmoscopy and photoreceptor inner segment (IS), outer segment (OS), and outer nuclear layer (ONL) thickness from optical coherence tomography (OCT) images. OCT angiography images were used to study choriocapillaris flow deficit percent (CCFD). Cone function was assessed by fundus-guided microperimetry. Measures were compared at designated regions using analysis of variance with pairwise comparisons among disease groups, adjusted for disease duration and eccentricity.

resultsOCT segmentation revealed shorter OS and IS, with reduced ONL thickness in RPGR compared to normal (OS: P < .001, IS: P = .001, ONL: P = .005), USH2 (OS: P = .01, IS: P = .03, ONL: P = .03), or ARRP (OS: P = .001, ONL: P = .03). Increased cone spacing was observed in both RPGR (P = .03) and USH2 compared with normal (P = .048). The mean CCFD in RPGR was greater than in USH2 (P = .02). Microperimetry demonstrated below-normal regional sensitivity in RPGR (P = .004), USH2 (P = .02), and ARRP (P = .009), without significant intergroup differences.

conclusionsOuter retinal structure and choriocapillaris perfusion were more abnormal in RPGR- than USH2A-related retinal degenerations, whereas there were no significant differences in below-normal regional sensitivity between each rod-cone degeneration associated with variants in these 2 genes expressed at the photoreceptor-connecting cilium.

Indexed as

Retinal DegenerationRetinitis PigmentosaUsher SyndromesCross-Sectional StudiesElectroretinographyExtracellular Matrix ProteinsEye ProteinsHumansRetinal Cone Photoreceptor CellsRetrospective StudiesTomography, Optical CoherenceExtracellular Matrix ProteinsEye ProteinsRPGR protein, humanUSH2A protein, human

Identifiers

PMID36646238
PMCPMC10308738
OpenAlexW4315929079

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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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.