Song Ni, Schwab Kristopher R, Patterson Larry T, Yamaguchi Terry, Lin Xinhua, Potter Steven S, Lang Richard A
Divisions of Pediatric Ophthalmology, Children's Hospital Research Foundation, Cincinnati, OH 45229, USA.
Development. 2007 May;134(10):1873-85. doi: 10.1242/dev.001495. Epub 2007 Apr 11.
Drosophila Pygopus was originally identified as a core component of the canonical Wnt signaling pathway and a transcriptional coactivator. Here we have investigated the microophthalmia that arises in mice with a germline null mutation of pygopus 2. We show that this phenotype is a consequence of defective lens development at inductive stages. Using a series of regionally limited Cre recombinase transgenes for conditional deletion of Pygo2(flox), we show that Pygo2 activity in pre-placodal presumptive lens ectoderm, placodal ectoderm and ocular mesenchyme all contribute to lens development. In each case, Pygo2 is required for normal expression levels of the crucial transcription factor Pax6. Finally, we provide multiple lines of evidence that although Pygo2 can function in the Wnt pathway, its activity in lens development is Wnt pathway-independent.
果蝇的Pygopus最初被鉴定为经典Wnt信号通路的核心成分和转录共激活因子。在此，我们研究了携带pygopus 2生殖系无效突变的小鼠中出现的小眼症。我们发现这种表型是诱导阶段晶状体发育缺陷的结果。使用一系列区域有限的Cre重组酶转基因对Pygo2（flox）进行条件性缺失，我们发现前基板假定晶状体外胚层、基板外胚层和眼间充质中的Pygo2活性均有助于晶状体发育。在每种情况下，Pygo2都是关键转录因子Pax6正常表达水平所必需的。最后，我们提供了多条证据表明，尽管Pygo2可以在Wnt通路中发挥作用，但其在晶状体发育中的活性不依赖于Wnt通路。
