Article
Tbx5 inhibits hedgehog signaling in determination of digit identity.
Human molecular genetics - 3 Jun 2020
Xu Huiting, Xiang Menglan, Qin Yushu, Cheng Henghui, Chen Duohua, Fu Qiang, Zhang Ke K, Xie Linglin
Abstract excerpt
Dominant TBX5 mutation causes Holt-Oram syndrome (HOS), which is characterized by limb defects in humans, but the underlying mechanistic basis is unclear. We used a mouse model with Tbx5 conditional knockdown in Hh-receiving cells (marked by Gli1+) during E8 to E10.5, a previously established model to study atrial septum defects, which displayed polydactyly or hypodactyly. The results suggested that Tbx5 is...
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