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Segmentation defects of Notch pathway mutants and absence of a synergistic phenotype in lunatic fringe/radical fringe double mutant mice
Author(s) -
Zhang Nian,
Norton Christine R.,
Gridley Thomas
Publication year - 2002
Publication title -
genesis
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.093
H-Index - 110
eISSN - 1526-968X
pISSN - 1526-954X
DOI - 10.1002/gene.10081
Subject(s) - mutant , notch signaling pathway , somitogenesis , phenotype , biology , microbiology and biotechnology , gene knockin , embryo , genetics , gene , signal transduction , embryogenesis , somite
Summary: 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. genesis 33:21–28, 2002. © 2002 Wiley‐Liss, Inc.