A tectorin-based matrix and planar-cell-polarity genes are required for normal collagen-fibril orientation in the developing tectorial membrane

Goodyear, Richard J, Lu, Xiaowei, Deans, Michael R and Richardson, Guy P (2017) A tectorin-based matrix and planar-cell-polarity genes are required for normal collagen-fibril orientation in the developing tectorial membrane. Development, 144. pp. 3978-3989. ISSN 0950-1991

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Abstract

The tectorial membrane is an extracellular structure of the cochlea. It develops on the surface of an epithelium and contains collagen fibrils embedded in a tectorin-based matrix. The collagen fibrils are oriented radially with an apically-directed slant - a feature considered critical for hearing. To determine how this pattern is generated, collagen-fibril formation was examined in mice lacking a tectorin-based matrix, epithelial cilia, or the planar-cell-polarity genes Vangl2 and Ptk7. In wild-type mice, collagen-fibril bundles appear within a tectorin-based matrix at E15.5 and, as fibril-number rapidly increases, become co-aligned and correctly oriented. Epithelial-width measurements and data from Kif3acKO mice suggest, respectively, radial stretch and cilia play little, if any, role in determining normal collagen-fibril orientation, but evidence from tectorin-knockout mice indicates confinement is important. PRICKLE2 distribution reveals the planar-cell-polarity axis in the underlying epithelium is organised along the length of the cochlea and, in mice in which this polarity is disrupted, the apically-directed collagen offset is no longer observed. These results highlight the importance of the tectorin-based matrix and epithelial signals for precise collagen organisation in the tectorial membrane

Item Type: Article
Keywords: Cochlea, Collagen, Hearing, Inner ear, Planar cell polarity, Tectorial membrane
Schools and Departments: School of Life Sciences > Neuroscience
Research Centres and Groups: Sussex Neuroscience
Depositing User: Guy Richardson
Date Deposited: 06 Dec 2017 09:42
Last Modified: 18 Jan 2018 12:36
URI: http://sro.sussex.ac.uk/id/eprint/71811

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Project NameSussex Project NumberFunderFunder Ref
The Tectorial Membrane and the Sensory Hair Bundles of the Inner Ear: Mechanisms of Development and Effects of Deafness-Causing MutationsG0162WELLCOME TRUST087737