Mouse Dach2 mutants do not exhibit gross defects in eye development or brain function

Genesis. 2006 Feb;44(2):84-92. doi: 10.1002/gene.20188.

Abstract

Drosophila dachshund is a critical regulator of eye, brain, and limb formation. Vertebrate homologs, Dach1 and Dach2, are expressed in the developing retina, brain, and limbs, suggesting functional conservation of the dachshund/Dach gene family. Dach1 mutants die postnatally, but exhibit grossly normal development. Here we report the generation of Dach2 mutant mice. Although deletion of Dach2 exon 1 results in abrogation of RNA expression, Dach2 mutants are viable and fertile. Histochemical analysis reveals grossly normal Dach2 mutant eye development. In addition, a battery of neurological assays failed to yield significant differences in behavior between Dach2 mutants and controls. We discuss these findings in the light of published observations of DACH2 mutations in the human population. Finally, to test the functional conservation hypothesis, we generated Dach2; Dach1 double mutant mice. Dach double mutants die after birth, similar to Dach1 homozygotes. However, unlike Drosophila dachshund mutants that lack eyes and exhibit leg truncations, the eyes and limbs of Dach double mutants are present, suggesting differences between Dach and dachshund gene function during embryonic eye and limb formation.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Brain / embryology
  • Brain / physiology*
  • DNA-Binding Proteins
  • Extremities / embryology
  • Eye / embryology
  • Eye / growth & development*
  • Eye Proteins / genetics
  • Female
  • Genes, X-Linked
  • Limb Deformities, Congenital / genetics
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Nuclear Proteins / deficiency
  • Nuclear Proteins / genetics*
  • Nuclear Proteins / physiology
  • Phenotype

Substances

  • DNA-Binding Proteins
  • Dach1 protein, mouse
  • Dach2 protein, mouse
  • Eye Proteins
  • Nuclear Proteins