Mice with an anterior cleft of the palate survive neonatal lethality

✍ Scribed by Shuping Gu; Na Wei; Xueyan Yu; Yiping Jiang; Jian Fei; YiPing Chen

• Publisher: John Wiley and Sons
• Year: 2008
• Tongue: English
• Weight: 713 KB
• Volume: 237
• Category: Article
• ISSN: 1058-8388
• DOI: 10.1002/dvdy.21534

✦ Synopsis

Abstract

Many genes are known to function in a region‐specific manner in the developing secondary palate. We have previously shown that Shox2‐deficient embryos die at mid‐gestation stage and develop an anterior clefting phenotype. Here, we show that mice carrying a conditional inactivation of Shox2 in the palatal mesenchyme survive the embryonic and neonatal lethality, but develop a wasting syndrome. Phenotypic analyses indicate a delayed closure of the secondary palate at the anterior end, leading to a failed fusion of the primary and secondary palates. Consistent with a role proposed for Shox2 in skeletogenesis, Shox2 inactivation causes a significantly reduced bone formation in the hard palate, probably due to a down‐regulation of Runx2 and Osterix. We conclude that the secondary palatal shelves are capable of fusion with each other, but fail to fuse with the primary palate in a developmentally delayed manner. Mice carrying an anterior cleft can survive neonatal lethality. Developmental Dynamics 237:1509‐1516, 2008. © 2008 Wiley‐Liss, Inc.