Cog4 is required for protrusion and extension of the epithelium in the developing semicircular canals. (February 2019)

• Record Type: Journal Article
• Authors: Clément, Aurélie; Blanco-Sánchez, Bernardo; Peirce, Judy L.; Westerfield, Monte
• Is Part Of: Mechanisms of development. Volume 155(2019)
• Page Start: 1
• Page End: 7
• Publication Date: 2019-02
• Subjects: Cog4 -- Pillar -- Semicircular canal -- Inner ear -- Zebrafish; Developmental biology -- Periodicals; Molecular biology -- Periodicals; Developmental Biology -- Periodicals; Molecular Biology -- Periodicals; Biologie du développement -- Périodiques; Biologie moléculaire -- Périodiques; Developmental biology; Molecular biology; Periodicals; Electronic journals; 571.8
• DOI: 10.1016/j.mod.2018.09.003
• Languages: English
• ISSNs: 0925-4773

Abstract

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. Highlights: Cog4 regulates development of the semicircular canals. cog4 is expressed in the early epithelial projections that fuse to form the semicircular canals. Growth of the epithelial projections is delayed in cog4 −/− homozygous zebrafish mutants. Cog4 regulates the secretion of extracellular matrix components required for protrusion and extension of epithelium.