Evidence map›Paper›PMID 37466006›Full record

ArticleDisease models & mechanisms2023

Ceramide compensation by ceramide synthases preserves retinal function and structure in a retinal dystrophy mouse model.

Xinye Qian, Tanmay Srinivasan, Jessica He, Jiaxiong Lu, Yan Jin, Haiwei Gu, Rui Chen

Open access · goldAbstract read
In one paragraph

Article in Disease models & mechanisms, 2023. 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
0.5field-weighted citation impact, top 33% 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, 3 citations in OpenAlex.

  1. Localization of Ceramide Synthase 5 in Mouse Retina.The Journal of comparative neurology · 2026
    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

7 authors at 3 institutions in 1 country.

Xinye QianVerna and Marrs McLean Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX 77030, USA.ORCID 0000-0003-1995-3621
Tanmay SrinivasanRice University, Houston, TX 77030, USA.
Jessica HeRice University, Houston, TX 77030, USA.
Jiaxiong LuVerna and Marrs McLean Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX 77030, USA.
Yan JinCenter for Translational Science, Florida International University, Port St. Lucie, FL 34987, USA.
Haiwei GuCenter for Translational Science, Florida International University, Port St. Lucie, FL 34987, USA.
Rui ChenHuman Genome Sequencing Center, Baylor College of Medicine, Houston, TX 77030, USA.ORCID 0000-0002-4387-9735
Baylor College of Medicine · USFlorida International University · USRice University · US

Funding

GENETICS OF EARLY ONSET RETINAL DISEASESR01EY018571 · NEI · UNIVERSITY OF CALIFORNIA-IRVINE · PI RUI CHEN · 2008 to 2026
$7.2M
Molecular Basis of Human Visual System DisordersR01EY022356 · NEI · UNIVERSITY OF CALIFORNIA-IRVINE · PI RUI CHEN · 2012 to 2026
$6.1M
Molecular Mechanisms of Human Retinal DiseaseR01EY020540 · NEI · BAYLOR COLLEGE OF MEDICINE · PI CHEN, RUI, MARDON, GRAEME · 2011 to 2017
$2.3M
NEI NIH HHS R01 EY018571NEI NIH HHS R01 EY020540NEI NIH HHS R01 EY022356
6 · The paper itself

Abstract

Increasing evidence has supported the role of ceramide as a mediator of photoreceptor dysfunction or cell death in ceramide accumulation and deficiency contexts. TLCD3B, a non-canonical ceramide synthase, was previously identified in addition to the six canonical ceramide synthases (CerSs), and the Tlcd3b-/- mouse model exhibited both retinal dysfunction and degeneration. As previous canonical CerS-deficient mouse models failed to display retinal degeneration, the mechanisms of how TLCD3B interacts with CerSs have not been investigated. Additionally, as the ceramide profile of each CerS is distinct, it is unclear whether the overall level or the homeostasis of different ceramide species plays a critical role in photoreceptor degeneration. Interactions between TLCD3B with canonical CerSs expressed in the retina were examined by subretinally injecting recombinant adeno-associated virus 8 vectors containing the Cers2 (rAAV8-CerS2), Cers4 (rAAV8-CerS4) and Cers5 (rAAV8-CerS5) genes. Injection of all three rAAV8-CerS vectors restored retinal functions as indicated by improved electroretinogram responses, but only rAAV8-CerS5 successfully retained retinal morphology in Tlcd3b-/- mice. CerSs and TLCD3B played partially redundant roles. Additionally, rather than acting as an integral entity, different ceramide species had different impacts on retinal cells, suggesting that the maintenance of the overall ceramide profile is critical for retinal function.

Indexed as

CeramidesRetinal DystrophiesAnimalsMiceOxidoreductasesRetinaCeramidesdihydroceramide desaturaseOxidoreductasesCeramideCeramide synthaseInherited retinal dystrophyRetinaTLCD3B

Identifiers

PMID37466006
PMCPMC10387349
OpenAlexW4384664684

What OpenQuestion holds

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LicenceCC BY
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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.