Abstract：
: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.
journal_name
Elifejournal_title
eLifeauthors
Bagwell J,Norman J,Ellis K,Peskin B,Hwang J,Ge X,Nguyen SV,McMenamin SK,Stainier DY,Bagnat Mdoi
10.7554/eLife.51221subject
Has Abstractpub_date
2020-01-29 00:00:00issn
2050-084Xpii
51221journal_volume
9pub_type
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