| RRC ID |
76500
|
| 著者 |
Bagwell J, Norman J, Ellis K, Peskin B, Hwang J, Ge X, Nguyen SV, McMenamin SK, Stainier DY, Bagnat M.
|
| タイトル |
Notochord vacuoles absorb compressive bone growth during zebrafish spine formation.
|
| ジャーナル |
Elife
|
| 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.
|
| 巻・号 |
9
|
| 公開日 |
2020-1-29
|
| DOI |
10.7554/eLife.51221
|
| PII |
51221
|
| PMID |
31995030
|
| PMC |
PMC7012607
|
| MeSH |
Animals
Gene Expression Regulation, Developmental
Mutation
Notochord / metabolism*
Receptor-Interacting Protein Serine-Threonine Kinases / genetics
Spine / growth & development*
Vacuoles / metabolism*
Zebrafish / embryology*
Zebrafish Proteins / genetics
|
| IF |
7.08
|
| リソース情報 |
| ゼブラフィッシュ |
SAGFF(LF)214A |
