Manley N R, Capecchi M R
Howard Hughes Medical Institute, Department of Human Genetics, University of Utah School of Medicine, Salt Lake City 84112, USA.
Dev Biol. 1997 Dec 15;192(2):274-88. doi: 10.1006/dbio.1997.8765.
Hox genes encode transcription factors that are used to regionalize the mammalian embryo. Analysis of mice carrying targeted mutations in individual and multiple Hox genes is beginning to reveal a complex network of interactions among these closely related genes which is responsible for directing the formation of spatially restricted tissues and structures. In this report we present an analysis of the genetic interactions between all members of the third paralogous group, Hoxa3, Hoxb3, and Hoxd3. Previous analysis has shown that although mice homozygous for loss-of-function mutations in either Hoxa3 or Hoxd3 have no defects in common, mice mutant for both genes demonstrate that these two genes strongly interact in a dosage-dependent manner. To complete the analysis of this paralogous gene family, mice with a targeted disruption of the Hoxb3 gene were generated. Homozygous mutants have minor defects at low penetrance in the formation of both the cervical vertebrae and the IXth cranial nerve. Analysis and comparison of all double-mutant combinations demonstrate that all three members of this paralogous group interact synergistically to affect the development of both neuronal and mesenchymal neural crest-derived structures, as well as somitic mesoderm-derived structures. Surprisingly, with respect to the formation of the cervical vertebrae, mice doubly mutant for Hoxa3 and Hoxd3 or Hoxb3 and Hoxd3 show an indistinguishable defect, loss of the entire atlas. This suggests that the identity of the specific Hox genes that are functional in a given region may not be as critical as the total number of Hox genes operating in that region.
Hox基因编码用于使哺乳动物胚胎区域化的转录因子。对携带单个和多个Hox基因靶向突变的小鼠进行的分析,开始揭示这些密切相关基因之间复杂的相互作用网络,该网络负责指导空间受限组织和结构的形成。在本报告中,我们对第三个旁系同源基因群的所有成员Hoxa3、Hoxb3和Hoxd3之间的遗传相互作用进行了分析。先前的分析表明,尽管Hoxa3或Hoxd3功能丧失突变的纯合小鼠没有共同的缺陷,但这两个基因的突变小鼠表明这两个基因以剂量依赖的方式强烈相互作用。为了完成对这个旁系同源基因家族的分析,我们构建了Hoxb3基因靶向破坏的小鼠。纯合突变体在颈椎和第九对脑神经形成过程中存在低外显率的轻微缺陷。对所有双突变组合的分析和比较表明, 这个旁系同源基因群的所有三个成员协同相互作用,影响神经嵴来源的神经元和间充质结构以及体节中胚层来源结构的发育。令人惊讶的是,就颈椎的形成而言,Hoxa3和Hoxd3或Hoxb3和Hoxd3的双突变小鼠表现出无法区分的缺陷,即整个寰椎缺失。这表明在给定区域中起作用的特定Hox基因的身份可能不如在该区域中起作用的Hox基因总数那么关键。