Evidence map›Paper›PMID 42554417›Full record

ArticleTranslational vision science & technology2026

Functional and Structural Characterization of a Large Animal Model of RDH5-Associated Retinopathy.

Laura M Ford, Laurence M Occelli, Mary E Enfield, Nastassia A Benjamin, Kelian Sun, Nate Pasmanter, Simon M Petersen-Jones

Abstract read
In one paragraph

Article in Translational vision science & technology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

7 authors.

Laura M FordGenetics and Genome Sciences, Michigan State University, East Lansing, MI, USA.
Laurence M OccelliDepartment of Small Animal Clinical Sciences, Michigan State University, East Lansing, MI, USA.
Mary E EnfieldDepartment of Small Animal Clinical Sciences, Michigan State University, East Lansing, MI, USA.
Nastassia A BenjaminDepartment of Small Animal Clinical Sciences, Michigan State University, East Lansing, MI, USA.
Kelian SunDepartment of Small Animal Clinical Sciences, Michigan State University, East Lansing, MI, USA.
Nate PasmanterDepartment of Small Animal Clinical Sciences, Michigan State University, East Lansing, MI, USA.
Simon M Petersen-JonesDepartment of Small Animal Clinical Sciences, Michigan State University, East Lansing, MI, USA.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Purpose: Inherited retinal diseases are a group of hereditary diseases that cause variable levels of blindness and affect a multitude of adults and children. One such disease is fundus albipunctatus (FA). FA is caused by autosomal recessive retinol dehydrogenase 5 (RDH5) mutations and results in rod dysfunction leading to night blindness and, in a subset of patients, macular degeneration (MD). We previously reported a spontaneous feline model of FA due to an RDH5 missense mutation. The affected cats showed rod dysfunction and a proportion developed degeneration of the area centralis (AC), equivalent to MD in humans. Methods: We used fundus confocal scanning laser ophthalmoscopy and spectral-domain optical coherence tomography imaging, six different electroretinography protocols, immunohistochemistry, and histology/transmission electron microscopy to further characterize this large-animal cat model. Results: In addition to rod dysfunction, cone recovery from intense stimulation was impaired. For RDH5-/- cats that developed AC degeneration, an initial elongation of rod outer segments with disorganization of the distal tips was initially detected in the AC and visual streak, suggestive of impaired shedding/phagocytosis. With progression, photoreceptors degenerated in the AC, matching the MD seen in some human patients. Conclusions: The RDH5-/- cat model recapitulates features of both rod and cone dysfunction seen in human patients with RDH5 mutations. Translational Relevance: The RDH5-/- cat model offers a unique opportunity to further understand the mechanisms of RDH5-associated retinopathies and to investigate potential therapeutic approaches.

Indexed as

Alcohol OxidoreductasesAnimalsCatsDisease Models, AnimalElectroretinographyHumansImmunohistochemistryMutation, MissenseOphthalmoscopyRetinal Cone Photoreceptor CellsRetinal DiseasesRetinal Rod Photoreceptor CellsTomography, Optical CoherenceAlcohol Oxidoreductasesretinol dehydrogenase 5

Identifiers

PMID42554417
PMCPMC13450645

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.