PUBLICATION

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

Authors
Söllner, C., Schwarz, H., Geisler, R., and Nicolson, T.
ID
ZDB-PUB-041014-4
Date
2004
Source
Development genes and evolution   214(12): 582-590 (Journal)
Registered Authors
Geisler, Robert, Nicolson, Teresa, Söllner, Christian
Keywords
Otolith biogenesis, Otopetrin 1, backstroke, Starmaker, Zebrafish
MeSH Terms
  • Amino Acid Sequence
  • Animals
  • Cloning, Molecular
  • Ear, Inner/cytology
  • Epithelium/chemistry
  • Epithelium/metabolism
  • Gene Expression/drug effects
  • Membrane Proteins/genetics
  • Membrane Proteins/physiology*
  • Molecular Sequence Data
  • Mutation
  • Oligodeoxyribonucleotides, Antisense/genetics
  • Oligodeoxyribonucleotides, Antisense/pharmacology
  • Otolithic Membrane/embryology*
  • Otolithic Membrane/metabolism
  • Protein Transport/genetics
  • Protein Transport/physiology
  • RNA, Messenger/analysis
  • RNA, Messenger/metabolism
  • Zebrafish/embryology*
  • Zebrafish/genetics
  • Zebrafish/physiology
  • Zebrafish Proteins/analysis*
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
  • Zebrafish Proteins/physiology*
PubMed
15480759 Full text @ Dev. Genes Evol.
Abstract
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.
Genes / Markers
Figures
Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes