Evidence map›Paper›PMID 38135239›Full record

ArticleAmerican journal of ophthalmology2024

Functional Vision in Patients With Biallelic USH2A Variants.

Elise Heon, Michele Melia, Laura E Bocchino, Lassana Samarakoon, Jacque L Duncan, Allison R Ayala, Isabelle Audo, Chris Bradley, Janet K Cheetham, Gislin Dagnelie and 7 more

Open access · greenAbstract readMulticenter Study
In one paragraph

Article in American journal of ophthalmology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.6field-weighted citation impact, top 29% 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

2 citing papers in PubMed, 4 citations in OpenAlex.

  1. Article
  2. Review
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

17 authors at 11 institutions in 4 countries.

Elise HeonFrom the Departments of Ophthalmology and Vision Sciences, The Hospital for Sick Children, The University of Toronto (E.H.), Toronto, Ontario, Canada.
Michele MeliaJaeb Center for Health Research (M.M., L.E.B., L.S., A.R.A.), Tampa, Florida, USA.
Laura E BocchinoJaeb Center for Health Research (M.M., L.E.B., L.S., A.R.A.), Tampa, Florida, USA.
Lassana SamarakoonJaeb Center for Health Research (M.M., L.E.B., L.S., A.R.A.), Tampa, Florida, USA.
Jacque L DuncanUniversity of California, San Francisco (J.L.D.), San Francisco, California, USA.
Allison R AyalaJaeb Center for Health Research (M.M., L.E.B., L.S., A.R.A.), Tampa, Florida, USA. Electronic address: ffbcorrespauth@jaeb.org.
Isabelle AudoInstitut de la Vision, Sorbonne Université, INSERM, CNRS (I.A.), Paris, France; Centre Hospitalier National d'Ophtalmologie des Quinze-Vingts, INSERM-DGOS CIC1423 (I.A.), Paris, France.
Chris BradleyWilmer Eye Institute, Johns Hopkins University (C.B., G.D.), Baltimore, Maryland, USA.
Janet K CheethamFoundation Fighting Blindness (J.K.C., T.A.D.), Columbia, Maryland, USA.
Gislin DagnelieWilmer Eye Institute, Johns Hopkins University (C.B., G.D.), Baltimore, Maryland, USA.
Todd A DurhamFoundation Fighting Blindness (J.K.C., T.A.D.), Columbia, Maryland, USA.
Carel B HoyngRadboud University Medical Center (C.B.H.), Nijmegen, the Netherlands.
Nieraj JainEmory Eye Center (N.J.), Atlanta, Georgia, USA.
Kanishka T JayasunderaKellogg Eye Center-University of Michigan (K.T.J.), Ann Arbor, Michigan, USA.
Mark E PennesiCasey Eye Institute-Oregon Health & Science University (M.E.P.), Portland, Oregon, USA.
Christina Y WengBaylor College of Medicine (C.Y.W.), Houston, Texas, USA.
Foundation Fighting Blindness Consortium Investigator Group
Jaeb Center for Health Research · USFoundation Fighting Blindness · USJohns Hopkins University · USBaylor College of Medicine · USCentre National de la Recherche Scientifique · FREmory Eye Center · USOregon Health & Science University · USRadboud University Nijmegen · NLUniversity of California, San Francisco · USUniversity of Toronto · CAW.K. Kellogg Foundation · US

Funding

Rapid-Prototyping and Design CoreP30EY002162 · NEI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI LAMBA, DEEPAK ASHOK · 1985 to 2024
$16.8M
Foundation Fighting Blindness RUSH2A - CONSORTIUM - 2016NEI NIH HHS P30 EY002162
6 · The paper itself

Abstract

purposeTo describe functional vision (FV) and investigate the relationship between FV, visual acuity (VA), and hill of vision (V

designMulticenter, international, cross-sectional study.

methodsIn individuals with biallelic disease-causing variants in USH2A, clinical diagnosis of Usher syndrome type 2 (USH2) or autosomal recessive nonsyndromic retinitis pigmentosa (ARRP) was based on history of hearing loss and audiology examinations. The VALVVFQ-48 was administered verbally to participants ≥18 years old. VA was measured in both eyes; V

resultsMedian age of 121 participants (76 with USH2, 45 with ARRP) was 41 years (range: 19-80); 54% were female. FV scores varied from -2.0 to 7.6 logits (median [interquartile range (IQR)]: 2.8 [1.5-3.8]). ARRP and USH2 participants had similar FV scores, both before [mean (95% CI): 2.8 (2.3-3.4) and 2.7 (2.3-3.2), respectively], and after [mean (95% CI): 2.5 (2.1-3.0) and 2.9 (2.6-3.3), respectively; P = .24] adjusting for age, VA, disease duration, and V

conclusionsBiallelic USH2A variants were associated with a large range of FV, yet similar in ARRP and USH2, despite hearing loss in USH2. The modified VALVVFQ-48 we evaluated is not ideal for detecting the impact of USH2A-associated retinal degenerations on activities of daily living.

Indexed as

Retinitis PigmentosaUsher SyndromesActivities of Daily LivingAdultAgedAged, 80 and overCross-Sectional StudiesExtracellular Matrix ProteinsFemaleHumansMaleMiddle AgedMutationYoung AdultExtracellular Matrix ProteinsUSH2A protein, human

Identifiers

PMID38135239
PMCPMC11844904
OpenAlexW4390031595

What OpenQuestion holds

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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.