1. Cog4 is required for protrusion and extension of the epithelium in the developing semicircular canals
- Author
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Bernardo Blanco-Sánchez, Judy L. Peirce, Monte Westerfield, and Aurélie Clément
- Subjects
Embryology ,Morphogenesis ,Article ,Epithelium ,Extracellular matrix ,03 medical and health sciences ,symbols.namesake ,0302 clinical medicine ,otorhinolaryngologic diseases ,medicine ,Animals ,Secretion ,Inner ear ,Hearing Loss ,Zebrafish ,030304 developmental biology ,0303 health sciences ,Hair Cells, Auditory, Inner ,biology ,Semicircular canal ,Gene Expression Regulation, Developmental ,Zebrafish Proteins ,Golgi apparatus ,biology.organism_classification ,Semicircular Canals ,Extracellular Matrix ,Cell biology ,medicine.anatomical_structure ,Mutation ,symbols ,sense organs ,030217 neurology & neurosurgery ,Developmental Biology - Abstract
The semicircular canals in the inner ear sense angular acceleration. In zebrafish, the semicircular canals develop from epithelial projections that grow toward each other and fuse to form pillars. The growth of the epithelial projections is driven by the production and secretion of extracellular matrix components by the epithelium. The conserved oligomeric Golgi 4 protein, Cog4, functions in retrograde vesicle transport within the Golgi and mutations can lead to sensory neural hearing loss. In zebrafish cog4 mutants, the inner ear is smaller and the number of hair cells is reduced. Here, we show that formation of the pillars is delayed and that secretion of extracellular matrix components (ECM) is impaired in cog4(−/−) mutants. These results show that Cog4 is required for secretion of ECM molecules essential to drive the growth of the epithelial projections and thus regulates morphogenesis of the semicircular canals.
- Published
- 2019
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