Segmentation defects of Notch pathway mutants and absence of a synergistic phenotype in lunatic fringe/radical fringe double mutant mice.
The Notch signaling pathway is important in regulating formation and anterior-posterior patterning of somites in vertebrate embryos. Here we show that distinct segmentation defects are displayed in embryos mutant for the Notch pathway genes Notch1, Lunatic fringe (Lfng), Delta-like 1 (Dll1), and Delta-like 3 (Dll3). Lfng-deficient mice and Dll3-deficient mice exhibit very similar defects, and marker analysis suggests that progression of the segmentation clock is disrupted in Dll3 mutants. We also show that Radical fringe (Rfng)-deficient mice exhibit no obvious phenotypic defects. To assess whether the absence of a phenotype in Rfng-deficient mice was the result of functional redundancy with the Lfng gene, we generated Lfng/Rfng double homozygous mutant mice. These mice exhibit the skeletal defects normally observed in Lfng-deficient mice, but we detected no obvious synergistic or additive effects in the double mutant animals.
Zhang
Nian
N
Van Andel Research Institute, Grand Rapids, Michigan, USA.
Norton
Christine R
CR
Gridley
Thomas
T
D000818:Animals; D005314:Embryonic and Fetal Development / Q000235:genetics; D005964:Glucosyltransferases; D016695:Glycosyltransferases / Q000235:genetics*; D006579:Heterozygote; D051379:Mice; D009154:Mutation; D010641:Phenotype; D011506:Proteins / Q000235:genetics*
D011506:Proteins; D016695:Glycosyltransferases; D005964:Glucosyltransferases; C498392:Lfng protein, mouse; C491317:Rfng protein, mouse