ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2020
PRPS polymerization influences lens fiber organization in zebrafish.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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Who cites it
10 citing papers in PubMed, 25 citations in OpenAlex.
- Inborn Errors of Purine Salvage and Catabolism.Metabolites · 2023Review
- Structural basis of human PRPS2 filaments.Cell & bioscience · 2023Article
- Greater than the sum of parts: Mechanisms of metabolic regulation by enzyme filaments.Current opinion in structural biology · 2023Review
- Human PRPS1 filaments stabilize allosteric sites to regulate activity.Nature structural & molecular biology · 2023Article
- CTP synthase: the hissing of the cellular serpent.Histochemistry and cell biology · 2022Review
- Molecular crowding facilitates bundling of IMPDH polymers and cytoophidium formation.Cellular and molecular life sciences : CMLS · 2022Article
- Article
- Review
- Towards Understanding PRPS1 as a Molecular Player in Immune Response in Yellow Drum (International journal of molecular sciences · 2022Article
- Review
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Authors and funding
3 authors at 2 institutions in 1 country.
Funding
Abstract
backgroundThe self-assembly of metabolic enzymes into filaments or foci highlights an intriguing mechanism for the regulation of metabolic activity. Recently, we identified the conserved polymerization of phosphoribosyl pyrophosphate synthetase (PRPS), which catalyzes the first step in purine nucleotide synthesis, in yeast and cultured mammalian cells. While previous work has revealed that loss of PRPS activity regulates retinal development in zebrafish, the extent to which PRPS filament formation affects tissue development remains unknown.
resultsBy generating novel alleles in the zebrafish PRPS paralogs, prps1a and prps1b, we gained new insight into the role of PRPS filaments during eye development. We found that mutations in prps1a alone are sufficient to generate abnormally small eyes along with defects in head size, pigmentation, and swim bladder inflation. Furthermore, a loss-of-function mutation that truncates the Prps1a protein resulted in the failure of PRPS filament assembly. Lastly, in mutants that fail to assemble PRPS filaments, we observed disorganization of the actin network in the lens fibers.
conclusionsThe truncation of Prps1a blocked PRPS filament formation and resulted in a disorganized lens fiber actin network. Altogether, these findings highlight a potential role for PRPS filaments during lens fiber organization in zebrafish.
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