Evidence map›Paper›PMID 34532411›Full record

ArticleAnnals of translational medicine2021

Retinal degeneration in mice and humans with neuronal ceroid lipofuscinosis type 8.

Elyse M Salpeter, Brian C Leonard, Antonio J Lopez, Christopher J Murphy, Sara Thomasy, Denise M Imai, Kristin Grimsrud, K C Kent Lloyd, Jiong Yan, Rossana Sanchez Russo and 2 more

Open access · diamondAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Annals of translational medicine, 2021. 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
0.4field-weighted citation impact, top 42% 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, 5 citations in OpenAlex.

  1. Article
  2. Clic6 Deficiency Triggers Aberrant Apical Microvilli in RPE.Investigative ophthalmology & visual science · 2025
    Article
  3. Review
  4. Advantages of ocular regeneration research.Annals of translational medicine · 2021
    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

12 authors at 2 institutions in 1 country.

Elyse M SalpeterWilliam R. Pritchard Veterinary Medical Teaching Hospital, School of Veterinary Medicine, University of California, Davis, Davis, California, USA.
Brian C LeonardWilliam R. Pritchard Veterinary Medical Teaching Hospital, School of Veterinary Medicine, University of California, Davis, Davis, California, USA.
Antonio J LopezDepartment of Ophthalmology & Vision Science, School of Medicine, University of California, Davis, Sacramento, California, USA.
Christopher J MurphyWilliam R. Pritchard Veterinary Medical Teaching Hospital, School of Veterinary Medicine, University of California, Davis, Davis, California, USA.
Sara ThomasyWilliam R. Pritchard Veterinary Medical Teaching Hospital, School of Veterinary Medicine, University of California, Davis, Davis, California, USA.
Denise M ImaiComparative Pathology Laboratory, School of Veterinary Medicine, UC Davis, Davis, California, USA.
Kristin GrimsrudMouse Biology Program, University of California, Davis, Davis, California, USA.
K C Kent LloydMouse Biology Program, University of California, Davis, Davis, California, USA.
Jiong YanDepartment of Ophthalmology, Emory University, Atlanta, Georgia, USA.
Rossana Sanchez RussoDepartment of Human Genetics, Emory University, Atlanta, Georgia, USA.
Suma P ShankarDepartment of Pediatrics & Department of Ophthalmology & Vision Science, School of Medicine, University of California, Davis, Sacramento, California, USA.
Ala MoshiriDepartment of Ophthalmology & Vision Science, School of Medicine, University of California, Davis, Sacramento, California, USA.
University of California, Davis · USEmory University · US

Funding

VISION RESEARCH CORE GRANTP30EY012576 · NEI · UNIVERSITY OF CALIFORNIA DAVIS · PI MARIE E BURNS · 1999 to 2026
$18.9M
The Role of ARAP1 in Retinal Photoreceptor HomeostasisK08EY027463 · NEI · UNIVERSITY OF CALIFORNIA AT DAVIS · PI MOSHIRI, ALA · 2017 to 2021
$1.0M
Characterizing the Impact of Genetic Polymorphism on Fentanyl Efficacy and Tolerance in PediatricsK01OD026608 · OD · UNIVERSITY OF CALIFORNIA AT DAVIS · PI GRIMSRUD, KRISTIN NICOLE · 2019 to 2023
$731k
NEI NIH HHS K08 EY027463NEI NIH HHS P30 EY012576NIH HHS K01 OD026608
6 · The paper itself

Abstract

backgroundCeroid lipofuscinosis type 8 belongs to a heterogenous group of vision and life-threatening neurodegenerative diseases, neuronal ceroid lipofuscinosis (NCL). Effective therapy is limited to a single drug for treatment of ceroid lipofuscinosis type 2, necessitating animal disease models to facilitate further therapeutic development. Murine models are advantageous for therapeutic development due to easy genetic manipulation and rapid breeding, however appropriate genetic models need to be identified and characterized before being used for therapy testing. To date, murine models of ocular disease associated with ceroid lipofuscinosis type 8 have only been characterized in motor neuron degeneration mice.

methods

resultsRetinal degeneration characterized by retinal pigment epithelium mottling, scattered drusen, and retinal vascular attenuation was noted in all

conclusionsThis study provides detailed clinical characterization of retinopathy in adult

Indexed as

murine modelNeuronal ceroid lipofuscinosisretinal degeneration

Identifiers

PMID34532411
PMCPMC8421982
OpenAlexW3153364459

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.