Polyketide Synthase Plays a Conserved Role in Otolith Formation

Mi Sun Lee, Julien Philippe, Elias Nicholas Katsanis, Weibin Zhou*

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

1 Scopus citations


Otoliths (ear stones) are biomineralized complexes essential for the balancing and hearing function of the inner ears in fish. Their formation is controlled by a genetically programmed biological process that is yet to be defined. We have isolated and characterized a spontaneous genetic mutant zebrafish with a complete absence of otoliths, named no otolith 1 (not1). not1 mutants are unable to develop otoliths during embryonic stages and fail to respond to acoustic stimuli, indicating an inner ear defect. We identified a deleterious mutation (G239R) that altered a highly conserved amino acid residue in the zebrafish ortholog of type I polyketide synthase (pks1) to underlie these phenotypes and showed that expression of the polyketide synthase gene of Japanese medaka fish could rescue the otolith deficiency in not1 mutant zebrafish. Our finding highlights a critical and conserved role of type I polyketide synthase in the initiation of otolith formation. Given the functional homology between otoliths in teleost fish and otoconia in mammals and humans, not1 mutants provide a new animal model for the study of human otoconia-related diseases.

Original languageEnglish (US)
Pages (from-to)363-369
Number of pages7
Issue number4
StatePublished - Aug 2019


  • Danio rerio
  • balancing
  • ear
  • hearing
  • otolith

ASJC Scopus subject areas

  • Animal Science and Zoology
  • Developmental Biology

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