ArticleThe Journal of neuroscience : the official journal of the Society for Neuroscience2010
Distinct retinal deficits in a zebrafish pyruvate dehydrogenase-deficient mutant.
Article in The Journal of neuroscience : the official journal of the Society for Neuroscience, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
What it found
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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.
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Who cites it
26 citing papers in PubMed, 38 citations in OpenAlex.
- Photoreceptor deletion of pyruvate dehydrogenase E1 subunit α1 induces retinal degeneration and reprograms retinal metabolism.Molecular metabolism · 2026Article
- Environmental Mixture Toxicity of Guanitoxin and Organophosphates in Zebrafish: from Developmental and Neurobehavioral Phenotypes to Transcriptomic Responses.Environmental science & technology · 2026Article
- Animal models of pyruvate dehydrogenase complex deficiency: insight into mechanisms of cerebral abnormalities and tissue-specific role in metabolism.Frontiers in medicine · 2026Review
- Modeling sacsin depletion in Danio Rerio offers new insight on retinal defects in ARSACS.Neurobiology of disease · 2025Article
- Review
- Comparing Ocular Toxicity of Legacy and Alternative Per- and Polyfluoroalkyl Substances in Zebrafish Larvae.Toxics · 2023Article
- Article
- Rods Contribute to Visual Behavior in Larval Zebrafish.Investigative ophthalmology & visual science · 2020Article
- A Great Catch for Investigating Inborn Errors of Metabolism-Insights Obtained from Zebrafish.Biomolecules · 2020Review
- ZebrafishThe Journal of neuroscience : the official journal of the Society for Neuroscience · 2020Article
- Fishing in the Cell Powerhouse: Zebrafish as A Tool for Exploration of Mitochondrial Defects Affecting the Nervous System.International journal of molecular sciences · 2019Review
- Article
- Reprogramming the metabolome rescues retinal degeneration.Cellular and molecular life sciences : CMLS · 2018Review
- A Brain-Derived Neurotrophic Factor Mimetic Is Sufficient to Restore Cone Photoreceptor Visual Function in an Inherited Blindness Model.Scientific reports · 2017Article
- Utilizing Zebrafish Visual Behaviors in Drug Screening for Retinal Degeneration.International journal of molecular sciences · 2017Review
- E4F1 controls a transcriptional program essential for pyruvate dehydrogenase activity.Proceedings of the National Academy of Sciences of the United States of America · 2016Article
- Wars2 is a determinant of angiogenesis.Nature communications · 2016Article
- Automated, high-throughput, in vivo analysis of visual function using the zebrafish.Developmental dynamics : an official publication of the American Association of Anatomists · 2016Article
- ATP6AP2/(pro)renin receptor contributes to glucose metabolism via stabilizing the pyruvate dehydrogenase E1 β subunit.The Journal of biological chemistry · 2015Article
- Probing Metabolism in the Intact Retina Using Stable Isotope Tracers.Methods in enzymology · 2015Review
Corrections and comments
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Authors and funding
4 authors at 1 institution in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Mutations in ubiquitously expressed metabolic genes often lead to CNS-specific effects, presumably because of the high metabolic demands of neurons. However, mutations in omnipresent metabolic pathways can conceivably also result in cell type-specific effects because of cell-specific requirements for intermediate products. One such example is the zebrafish noir mutant, which we found to be mutated in the pdhb gene, coding for the E1 beta subunit of the pyruvate dehydrogenase complex. This vision mutant is described as blind and was isolated because of its vision defect-related darker appearance. A detailed morphological, behavioral, and physiological analysis of the phenotype revealed an unexpected specific effect on the retina. Surprisingly, the cholinergic amacrine cells of the inner retina are affected earlier than the photoreceptors. This might be attributable to the inability of these cells to maintain production of their neurotransmitter acetylcholine. This is reflected in an earlier loss of motion vision, followed only later by a general loss of light perception. Since both characteristics of the phenotype are attributable to a loss of acetyl-CoA production by pyruvate dehydrogenase, we used a ketogenic diet to bypass this metabolic block and could indeed partially rescue vision and prolong survival of the larvae. The noir mutant provides a case for a systemic disease with ocular manifestation with a surprising specific effect on the retina given the ubiquitous requirement for the mutated gene.
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Registered trials
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.