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Notochord vacuoles absorb compressive bone growth during zebrafish spine formation.

Publication ,  Journal Article
Bagwell, J; Norman, J; Ellis, K; Peskin, B; Hwang, J; Ge, X; Nguyen, SV; McMenamin, SK; Stainier, DY; Bagnat, M
Published in: Elife
January 29, 2020

The vertebral column or spine assembles around the notochord rod which contains a core made of large vacuolated cells. Each vacuolated cell possesses a single fluid-filled vacuole, and loss or fragmentation of these vacuoles in zebrafish leads to spine kinking. Here, we identified a mutation in the kinase gene dstyk that causes fragmentation of notochord vacuoles and a severe congenital scoliosis-like phenotype in zebrafish. Live imaging revealed that Dstyk regulates fusion of membranes with the vacuole. We find that localized disruption of notochord vacuoles causes vertebral malformation and curving of the spine axis at those sites. Accordingly, in dstyk mutants the spine curves increasingly over time as vertebral bone formation compresses the notochord asymmetrically, causing vertebral malformations and kinking of the axis. Together, our data show that notochord vacuoles function as a hydrostatic scaffold that guides symmetrical growth of vertebrae and spine formation.

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Published In

Elife

DOI

EISSN

2050-084X

Publication Date

January 29, 2020

Volume

9

Location

England

Related Subject Headings

  • Zebrafish Proteins
  • Zebrafish
  • Vacuoles
  • Spine
  • Receptor-Interacting Protein Serine-Threonine Kinases
  • Notochord
  • Mutation
  • Gene Expression Regulation, Developmental
  • Animals
  • 42 Health sciences
 

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Bagwell, J., Norman, J., Ellis, K., Peskin, B., Hwang, J., Ge, X., … Bagnat, M. (2020). Notochord vacuoles absorb compressive bone growth during zebrafish spine formation. Elife, 9. https://doi.org/10.7554/eLife.51221
Bagwell, Jennifer, James Norman, Kathryn Ellis, Brianna Peskin, James Hwang, Xiaoyan Ge, Stacy V. Nguyen, Sarah K. McMenamin, Didier Yr Stainier, and Michel Bagnat. “Notochord vacuoles absorb compressive bone growth during zebrafish spine formation.Elife 9 (January 29, 2020). https://doi.org/10.7554/eLife.51221.
Bagwell J, Norman J, Ellis K, Peskin B, Hwang J, Ge X, et al. Notochord vacuoles absorb compressive bone growth during zebrafish spine formation. Elife. 2020 Jan 29;9.
Bagwell, Jennifer, et al. “Notochord vacuoles absorb compressive bone growth during zebrafish spine formation.Elife, vol. 9, Jan. 2020. Pubmed, doi:10.7554/eLife.51221.
Bagwell J, Norman J, Ellis K, Peskin B, Hwang J, Ge X, Nguyen SV, McMenamin SK, Stainier DY, Bagnat M. Notochord vacuoles absorb compressive bone growth during zebrafish spine formation. Elife. 2020 Jan 29;9.

Published In

Elife

DOI

EISSN

2050-084X

Publication Date

January 29, 2020

Volume

9

Location

England

Related Subject Headings

  • Zebrafish Proteins
  • Zebrafish
  • Vacuoles
  • Spine
  • Receptor-Interacting Protein Serine-Threonine Kinases
  • Notochord
  • Mutation
  • Gene Expression Regulation, Developmental
  • Animals
  • 42 Health sciences