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

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posted on 2023-06-09, 09:11 authored by Richard GoodyearRichard Goodyear, Xiaowei Lu, Michael R Deans, Guy Richardson
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

Funding

The Tectorial Membrane and the Sensory Hair Bundles of the Inner Ear: Mechanisms of Development and Effects of Deafness-Causing Mutations; G0162; WELLCOME TRUST; 087737

History

Publication status

  • Published

File Version

  • Accepted version

Journal

Development

ISSN

0950-1991

Publisher

The company of biologists

Volume

144

Page range

3978-3989

Department affiliated with

  • Neuroscience Publications

Research groups affiliated with

  • Sussex Neuroscience Publications

Full text available

  • Yes

Peer reviewed?

  • Yes

Legacy Posted Date

2017-12-06

First Open Access (FOA) Date

2018-09-21

First Compliant Deposit (FCD) Date

2017-12-06

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