ArticleMechanisms of development2019
Zebrafish otolith biomineralization requires polyketide synthase.
Article in Mechanisms of development, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.
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Who cites it
11 citing papers in PubMed, 16 citations in OpenAlex.
- AI-assisted phenotyping in a zebrafish hypophosphatasia model enables early and precise detection of skeletal alterations.Scientific reports · 2025Article
- Development of the semicircular canals and otolithic organs of the vertebrate inner ear.Current topics in developmental biology · 2025Review
- Bdelloid rotifers deploy horizontally acquired biosynthetic genes against a fungal pathogen.Nature communications · 2024Article
- Metabonomics and Transcriptomics Analyses Reveal the Development Process of the Auditory System in the Embryonic Development Period of the Small Yellow Croaker under Background Noise.International journal of molecular sciences · 2024Article
- Sea Urchin Polyketide Synthase SpPks1 Produces the Naphthalene Precursor to Echinoderm Pigments.Journal of the American Chemical Society · 2022Article
- Advances in otolith-related protein research.Frontiers in neuroscience · 2022Review
- A genetic modifier of venous thrombosis in zebrafish reveals a functional role for fibrinogen AαE in early hemostasis.Blood advances · 2020Article
- Cilia in the developing zebrafish ear.Philosophical transactions of the Royal Society of London. Series B, Biological sciences · 2020Review
- Linking Genes to Molecules in Eukaryotic Sources: An Endeavor to Expand Our Biosynthetic Repertoire.Molecules (Basel, Switzerland) · 2020Review
- The biosynthetic diversity of the animal world.The Journal of biological chemistry · 2019Review
- Polyketide Synthase Plays a Conserved Role in Otolith Formation.Zebrafish · 2019Article
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Authors and funding
7 authors at 3 institutions in 2 countries.
Funding
Abstract
Deflecting biomineralized crystals attached to vestibular hair cells are necessary for maintaining balance. Zebrafish (Danio rerio) are useful organisms to study these biomineralized crystals called otoliths, as many required genes are homologous to human otoconial development. We sought to identify and characterize the causative gene in a trio of homozygous recessive mutants, no content (nco) and corkscrew (csr), and vanished (vns), which fail to develop otoliths during early ear development. We show that nco, csr, and vns have potentially deleterious mutations in polyketide synthase (pks1), a multi-modular protein that has been previously implicated in biomineralization events in chordates and echinoderms. We found that Otoconin-90 (Oc90) expression within the otocyst is diffuse in nco and csr; therefore, it is not sufficient for otolith biomineralization in zebrafish. Similarly, normal localization of Otogelin, a protein required for otolith tethering in the otolithic membrane, is not sufficient for Oc90 attachment. Furthermore, eNOS signaling and Endothelin-1 signaling were the most up- and down-regulated pathways during otolith agenesis in nco, respectively. Our results demonstrate distinct processes for otolith nucleation and biomineralization in vertebrates and will be a starting point for models that are independent of Oc90-mediated seeding. This study will serve as a basis for investigating the role of eNOS signaling and Endothelin-1 signaling during otolith formation.
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