Mutated otopetrin 1 affects the genesis of otoliths and the localization of Starmaker in zebrafish

Christian Söllner, Heinz Schwarz, Robert Geisler, Teresa Nicolson

Research output: Contribution to journalArticlepeer-review

59 Scopus citations


Otoliths in bony fishes and otoconia in mammals are composite crystals consisting of calcium carbonate and proteins. These biominerals are part of the gravity and linear acceleration detection system of the inner ear. Mutations in otopetrin 1 have been shown to result in lack of otoconia in tilted and mergulhador mutant mice. The molecular function of Otopetrin 1, a novel protein that contains ten predicted transmembrane domains, however, has remained elusive. Here we show that a mutation in the orthologous gene in zebrafish is responsible for the complete absence of otoliths in backstroke mutants. We examined the localization of Starmaker, a secreted protein that is highly abundant in otoliths in backstroke mutants. Starmaker protein accumulated within cells of the otic epithelium, indicating a possible defect in secretion. Our data suggest that Otopetrin 1 in zebrafish may be involved in the protein trafficking of components required for formation of biominerals in the ear.

Original languageEnglish (US)
Pages (from-to)582-590
Number of pages9
JournalDevelopment Genes and Evolution
Issue number12
StatePublished - Dec 2004
Externally publishedYes


  • Backstroke
  • Otolith biogenesis
  • Otopetrin 1
  • Starmaker
  • Zebrafish

ASJC Scopus subject areas

  • Genetics
  • Developmental Biology


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