Loss of the Tg737 protein results in skeletal patterning defects.
Document Type
Article
Publication Date
2003
First Page
78
Last Page
90
JAX Source
Dev Dyn 2003 May; 227(1):78-90.
Abstract
Tg737 mutant mice exhibit pathologic conditions in numerous tissues along with skeletal patterning defects. Herein, we characterize the skeletal pathologic conditions and confirm a role for Tg737 in skeletal patterning through transgenic rescue. Analyses were conducted in both the hypomorphic Tg737(orpk) allele that results in duplication of digit one and in the null Tg737(Delta2-3betaGal) allele that is an embryonic lethal mutation exhibiting eight digits per limb. In early limb buds, Tg737 expression is detected throughout the mesenchyme becoming concentrated in precartilage condensations at later stages. In situ analyses indicate that the Tg737(orpk) mutant limb defects are not associated with changes in expression of Shh, Ihh, HoxD11-13, Patched, BMPs, or Glis. Likewise, in Tg737(Delta2-3betaGal) mutant embryos, there was no change in Shh expression. However, in both alleles, Fgf4 was ectopically expressed on the anterior apical ectodermal ridge. Collectively, the data argue for a dosage effect of Tg737 on the limb phenotypes and that the polydactyly is independent of Shh misexpression. Developmental Dynamics 227:78-90, 2003.
Recommended Citation
Zhang Q,
Murcia NS,
Chittenden LR,
Richards WG,
Michaud EJ,
Woychik RP,
Yoder BK.
Loss of the Tg737 protein results in skeletal patterning defects. Dev Dyn 2003 May; 227(1):78-90.