Broad Center for Regenerative Medicine and Stem Cell Research, University of Southern California Keck School of Medicine, 1425 San Pablo St., Los Angeles, CA 90033, USA.
Dev Biol. 2012 Jan 1;361(1):116-24. doi: 10.1016/j.ydbio.2011.10.018. Epub 2011 Oct 20.
Disruption of retinoic acid signaling causes a variety of pharyngeal arch artery and great vessel defects, as well as malformations in many other tissues, including those derived from the pharyngeal endoderm. Previous studies implied that arch artery defects in the context of defective RA signaling occur secondary to pharyngeal pouch segmentation defects, although this model has never been experimentally verified. In this study, we examined arch artery morphogenesis during mouse development, and the role of RA in this process. We show in normal embryos that the arch arteries form by vasculogenic differentiation of pharyngeal mesoderm. Using various genetic backgrounds and tissue-specific mutation approaches, we segregate pharyngeal arch artery and pharyngeal pouch defects in RA receptor mutants, and show that RA signal transduction only in pharyngeal mesoderm is required for arch artery formation. RA does not control pharyngeal mesodermal differentiation to endothelium, but instead promotes the aggregation of endothelial cells into nascent vessels. Expression of VE-cadherin was substantially reduced in RAR mutants, and this deficiency may underlie the arch artery defects. The consequences of disrupted mesodermal and endodermal RA signaling were restricted to the 4th and 6th arch arteries and to the 4th pharyngeal pouch, respectively, suggesting that different regulatory mechanisms control the formation of the more anterior arch arteries and pouches.
视黄酸信号通路的破坏会导致多种咽弓动脉和大血管缺陷,以及许多其他组织的畸形,包括咽内胚层衍生的组织。先前的研究表明,在视黄酸信号转导缺陷的情况下,弓动脉缺陷是由于咽囊分段缺陷引起的,尽管这种模型从未经过实验验证。在这项研究中,我们检查了小鼠发育过程中的弓动脉形态发生,以及 RA 在这个过程中的作用。我们在正常胚胎中显示,弓动脉通过咽中胚层的血管生成分化形成。使用各种遗传背景和组织特异性突变方法,我们在 RA 受体突变体中分离出弓动脉和咽囊缺陷,并表明 RA 信号转导仅在咽中胚层中是弓动脉形成所必需的。RA 不控制咽中胚层向内皮细胞的分化,而是促进内皮细胞聚集形成新生血管。在 RAR 突变体中,VE-钙黏蛋白的表达显著降低,这可能是弓动脉缺陷的基础。中胚层和内胚层 RA 信号通路的破坏的后果仅限于第 4 和第 6 对咽弓动脉和第 4 对咽囊,这表明不同的调节机制控制着更靠前的咽弓动脉和咽囊的形成。