Patterns & Phenotypes
Tulp3 is a critical repressor of mouse hedgehog signaling
Article first published online: 30 MAR 2009
Copyright © 2009 Wiley-Liss, Inc.
Volume 238, Issue 5, pages 1140–1149, May 2009
How to Cite
Cameron, D. A., Pennimpede, T. and Petkovich, M. (2009), Tulp3 is a critical repressor of mouse hedgehog signaling. Dev. Dyn., 238: 1140–1149. doi: 10.1002/dvdy.21926
- Issue published online: 21 APR 2009
- Article first published online: 30 MAR 2009
- Manuscript Accepted: 14 FEB 2009
- Canadian Institutes of Health. Grant Number: 394363
- National Cancer Institute of Canada. Grant Number: 366098
- sonic hedgehog;
- neural patterning;
- limb development
Precise regulation of the morphogen sonic hedgehog (Shh) and modulation of the Shh signaling pathway is required for proper specification of cell fate within the developing limbs and neural tube, and resultant tissue morphogenesis. Tulp3 (tubby-like protein 3) is a protein of unknown function which has been implicated in nervous system development through gene knockout studies. We demonstrate here that mice lacking the Tulp3 gene develop abnormalities of both the neural tube and limbs consistent with improper regulation of Shh signaling. Tulp3−/− embryos show expansion of Shh target gene expression and display a ventralization of neural progenitor cells in the caudal neural tube. We further show that Tulp3−/−/Shh−/− compound mutant embryos resemble Tulp3 mutants, and express Shh target genes in the neural tube and limbs which are not expressed in Shh−/− embryos. This work uncovers a novel role for Tulp3 as a negative regulatory factor in the Hh pathway. Developmental Dynamics 238:1140–1149, 2009. © 2009 Wiley-Liss, Inc.