Journal
GENESIS
Volume 53, Issue 9, Pages 573-582Publisher
WILEY-BLACKWELL
DOI: 10.1002/dvg.22875
Keywords
ENU; mutagenesis; forebrain; development; mouse; cloning
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Funding
- National Institutes of Health [HD36404, MH081187, HG0003985, NS085023]
- Cincinnati Children's Hospital Research Foundation
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Grainyhead-like genes are part of a highly conserved gene family that play a number of roles in ectoderm development and maintenance in mammals. Here we identify a novel allele of Grhl2, cleft-face 3 (clft3), in a mouse line recovered from an ENU mutagenesis screen for organogenesis defects. Homozygous clft3 mutants have a number of phenotypes in common with other alleles of Grhl2. We note a significant effect of genetic background on the clft3 phenotype. One of these is a reduction in size of the telencephalon where we find abnormal patterns of neural progenitor mitosis and apoptosis in mutant brains. Interestingly, Grhl2 is not expressed in the developing forebrain, suggesting this is a survival factor for neural progenitors exerting a paracrine effect on the neural tissue from the overlying ectoderm where Grhl2 is highly expressed. (C) 2015 Wiley Periodicals, Inc.
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