ZFIN ID: ZDB-PUB-140816-8
Understanding cardiac sarcomere assembly with zebrafish genetics
Yang, J., Shih, Y.H., Xu, X.
Date: 2014
Source: Anatomical record (Hoboken, N.J. : 2007)   297: 1681-93 (Review)
Registered Authors: Shih, Yu-huan, Xu, Xiaolei, Yang, Jingchun
Keywords: cardiomyopathy, genetics, myofibrillogenesis, sarcomere, zebrafish
MeSH Terms:
  • Animals
  • Animals, Genetically Modified
  • Genetic Predisposition to Disease
  • Genotype
  • Heart Diseases/genetics
  • Heart Diseases/metabolism
  • Heart Diseases/pathology
  • Humans
  • Mutation
  • Myocytes, Cardiac/metabolism*
  • Myocytes, Cardiac/pathology
  • Phenotype
  • Risk Factors
  • Sarcomeres/metabolism*
  • Sarcomeres/pathology
  • Zebrafish/genetics*
  • Zebrafish/metabolism
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/metabolism
PubMed: 25125181 Full text @ Anat. Rec. (Hoboken)
Mutations in sarcomere genes have been found in many inheritable human diseases, including hypertrophic cardiomyopathy. Elucidating the molecular mechanisms of sarcomere assembly shall facilitate understanding of the pathogenesis of sarcomere-based cardiac disease. Recently, biochemical and genomic studies have identified many new genes encoding proteins that localize to the sarcomere. However, their precise functions in sarcomere assembly and sarcomere-based cardiac disease are unknown. Here, we review zebrafish as an emerging vertebrate model for these studies. We summarize the techniques offered by this animal model to manipulate genes of interest, annotate gene expression, and describe the resulting phenotypes. We survey the sarcomere genes that have been investigated in zebrafish and discuss the potential of applying this in vivo model for larger-scale genetic studies.