ZFIN ID: ZDB-PUB-180418-14
The DEAD box RNA helicase Ddx39ab is essential for myocyte and lens development in zebrafish
Zhang, L., Yang, Y., Li, B., Scott, I.C., Lou, X.
Date: 2018
Source: Development (Cambridge, England)   145(8): (Journal)
Registered Authors: Li, Beibei, Lou, Xin, Scott, Ian, Yang, Yuxi, Zhang, Linlin
Keywords: Lens, Myocyte, RNA helicase, RNA splicing, Zebrafish
MeSH Terms:
  • Animals
  • Animals, Genetically Modified
  • Cell Differentiation/genetics
  • Cell Lineage/genetics
  • DEAD-box RNA Helicases/genetics*
  • DEAD-box RNA Helicases/metabolism
  • Epigenesis, Genetic
  • Gene Expression Regulation, Developmental
  • Gene Expression Regulation, Enzymologic
  • Gene Knockout Techniques
  • Histone-Lysine N-Methyltransferase/genetics
  • Histone-Lysine N-Methyltransferase/metabolism
  • Lens, Crystalline/embryology
  • Lens, Crystalline/metabolism
  • Muscle Cells/cytology
  • Muscle Cells/metabolism
  • Mutation
  • Phenotype
  • RNA Splicing
  • Zebrafish/embryology*
  • Zebrafish/genetics*
  • Zebrafish/metabolism
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/metabolism
PubMed: 29636379 Full text @ Development
RNA helicases from the DEAD-box family are found in almost all organisms and have important roles in RNA metabolism, including RNA synthesis, processing and degradation. The function and mechanism of action of most of these helicases in animal development and human disease remain largely unexplored. In a zebrafish mutagenesis screen to identify genes essential for heart development we identified a mutant that disrupts the gene encoding the RNA helicase DEAD-box 39ab (ddx39ab). Homozygous ddx39ab mutant embryos exhibit profound cardiac and trunk muscle dystrophy, along with lens abnormalities, caused by abrupt terminal differentiation of cardiomyocyte, myoblast and lens fiber cells. Loss of ddx39ab hindered splicing of mRNAs encoding epigenetic regulatory factors, including members of the KMT2 gene family, leading to misregulation of structural gene expression in cardiomyocyte, myoblast and lens fiber cells. Taken together, these results show that Ddx39ab plays an essential role in establishment of the proper epigenetic status during differentiation of multiple cell lineages.