A simple strategy for heritable chromosomal deletions in zebrafish via the combinatorial action of targeting nucleases

Genome Biol. 2013 Jul 1;14(7):R69. doi: 10.1186/gb-2013-14-7-r69.


Precise and effective genome-editing tools are essential for functional genomics and gene therapy. Targeting nucleases have been successfully used to edit genomes. However, whole-locus or element-specific deletions abolishing transcript expression have not previously been reported. Here, we show heritable targeting of locus-specific deletions in the zebrafish nodal-related genes squint (sqt) and cyclops (cyc). Our strategy of heritable chromosomal editing can be used for disease modeling, analyzing gene clusters, regulatory regions, and determining the functions of non-coding RNAs in genomes.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Alleles
  • Animals
  • Base Sequence
  • Chromosome Deletion*
  • Crosses, Genetic
  • Deoxyribonucleases / metabolism*
  • Embryo, Nonmammalian / metabolism
  • Female
  • Genetic Loci
  • Genomics / methods*
  • Germ-Line Mutation / genetics
  • Green Fluorescent Proteins / metabolism
  • Inheritance Patterns / genetics*
  • Male
  • Molecular Sequence Data
  • RNA / genetics
  • Zebrafish / embryology
  • Zebrafish / genetics*
  • Zinc Fingers / genetics


  • enhanced green fluorescent protein
  • Green Fluorescent Proteins
  • RNA
  • Deoxyribonucleases