PUBLICATION

The axonemal dynein heavy chain 10 gene is essential for monocilia motility and spine alignment in zebrafish

Authors
Wang, Y., Troutwine, B.R., Zhang, H., Gray, R.S.
ID
ZDB-PUB-211217-15
Date
2021
Source
Developmental Biology   482: 82-90 (Journal)
Registered Authors
Gray, Ryan, Troutwine, Benjamin
Keywords
Dynein, Motile cilia, Reissner fiber, Scoliosis
MeSH Terms
  • Animals
  • Axonemal Dyneins/genetics*
  • Cell Movement/genetics
  • Cilia/genetics*
  • Cilia/metabolism
  • Disease Models, Animal
  • Morphogenesis/genetics
  • Scoliosis/genetics*
  • Scoliosis/physiopathology
  • Spine/embryology*
  • Spine/physiology
  • Zebrafish/embryology*
  • Zebrafish/genetics
  • Zebrafish Proteins/genetics
PubMed
34915022 Full text @ Dev. Biol.
Abstract
Adolescent idiopathic scoliosis (AIS) is a common pediatric musculoskeletal disorder worldwide, characterized by atypical spine curvatures in otherwise healthy children. Human genetic studies have identified candidate genes associated with AIS, however, only a few of these have been shown to recapitulate adult-viable scoliosis in animal models. Using an F0 CRISPR screening approach in zebrafish, we demonstrate that disruption of the dynein axonemal heavy chain 10 (dnah10) gene results in recessive adult-viable scoliosis in zebrafish. Using a stably segregating dnah10 mutant zebrafish, we showed that the ependymal monocilia lining the hindbrain and spinal canal displayed reduced beat frequency, which was correlated with the disassembly of the Reissner fiber and the onset of body curvatures. Taken together, these results suggest that monocilia function in larval zebrafish contributes to the polymerization of the Reissner fiber and straightening of the body axis.
Genes / Markers
Figures
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Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes