Notochord vacuoles absorb compressive bone growth during zebrafish spine formation.

Journal Article (Journal Article)

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.

Full Text

Duke Authors

Cited Authors

  • Bagwell, J; Norman, J; Ellis, K; Peskin, B; Hwang, J; Ge, X; Nguyen, SV; McMenamin, SK; Stainier, DY; Bagnat, M

Published Date

  • January 29, 2020

Published In

Volume / Issue

  • 9 /

PubMed ID

  • 31995030

Pubmed Central ID

  • PMC7012607

Electronic International Standard Serial Number (EISSN)

  • 2050-084X

Digital Object Identifier (DOI)

  • 10.7554/eLife.51221


  • eng

Conference Location

  • England