Patterns & Phenotypes

You have full text access to this OnlineOpen article

Tulp3 is a critical repressor of mouse hedgehog signaling

  1. Don A. Cameron1,2,
  2. Tracie Pennimpede2,3,
  3. Martin Petkovich1,2,3,*

Article first published online: 30 MAR 2009

DOI: 10.1002/dvdy.21926

Issue

Developmental Dynamics

Developmental Dynamics

Volume 238, Issue 5, pages 1140–1149, May 2009

Additional Information

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

Author Information

  1. 1

    Department of Biochemistry, Cancer Research Institute, Queen's University, Kingston, Ontario, Canada

  2. 2

    Division of Cancer Biology and Genetics, Cancer Research Institute, Queen's University, Kingston, Ontario, Canada

  3. 3

    Department of Pathology and Molecular Medicine, Cancer Research Institute, Queen's University, Kingston, Ontario, Canada

*Division of Cancer Biology and Genetics, Cancer Research Institute, Queen's University, Botterell Hall Room 355, Kingston, ON, Canada, K7L 3N6

Publication History

  1. Issue published online: 21 APR 2009
  2. Article first published online: 30 MAR 2009
  3. Manuscript Accepted: 14 FEB 2009

Funded by

  • Canadian Institutes of Health. Grant Number: 394363
  • National Cancer Institute of Canada. Grant Number: 366098

SEARCH

SEARCH BY CITATION

ARTICLE TOOLS

View Full Article (HTML) Get PDF (10517K)

Keywords:

  • Tulp3;
  • sonic hedgehog;
  • neural patterning;
  • limb development

Abstract

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.

View Full Article (HTML) Get PDF (10517K)