Brandt William, Khandekar Melin, Suzuki Norio, Yamamoto Masayuki, Lim Kim-Chew, Engel James Douglas
Department of Cell and Developmental Biology, University of Michigan Medical School, 109 Zina Pitcher Place, Ann Arbor, MI 48109-2200, USA.
J Biol Chem. 2008 Apr 4;283(14):8976-83. doi: 10.1074/jbc.M709364200. Epub 2008 Jan 21.
Transcription factor GATA-2 is vital for both hematopoietic progenitor cell function and urogenital patterning. Transgenic mapping studies have shown that the hematopoietic and urogenital enhancers are located hundreds of kbp 5' and 3' to the Gata2 structural gene, and both are vital for embryonic development. Because the size of mammalian genes, including all of their associated regulatory elements, can exceed a megabase, transgenic complementation in mice has, in specific instances, proven to be a formidable hurdle. After incorporating the Gata2 structural gene as well as the distant hematopoietic and urogenital enhancers into a single, contiguous piece of DNA by fusing two bacterial artificial chromosomes (BACs) into one, we formally tested the hypothesis that the functional boundaries of this locus are contained within this contiguous genomic span. We show that two independent lines of transgenic mice bearing a multicopy 413-kbp-linked Gata2 BAC transgene (bearing sequences from -187 to +226 kbp of the locus) are able to fully rescue Gata2 null mutant embryonic lethality and that the rescued animals behave and reproduce normally. Surprisingly, the linked BAC confers expression in the ureteric epithelium, whereas sequences within any of the overlapping parental BACs and a yeast artificial chromosome that were originally tested do not, and thus these experiments also define a novel synthetic enhancer activity that has not been previously described. These genetic complementation studies define the required outer limits of the Gata2 locus and formally demonstrate that enhancers lying beyond those boundaries are not necessary for Gata2-regulated viability or fecundity.
转录因子GATA-2对造血祖细胞功能和泌尿生殖系统模式形成都至关重要。转基因定位研究表明,造血和泌尿生殖增强子位于Gata2结构基因5'端和3'端数百千碱基对处,两者对胚胎发育都至关重要。由于包括所有相关调控元件在内的哺乳动物基因大小可能超过1兆碱基,在特定情况下,小鼠中的转基因互补已被证明是一个巨大的障碍。通过将两个细菌人工染色体(BAC)融合成一个,将Gata2结构基因以及远处的造血和泌尿生殖增强子整合到一段连续的DNA中后,我们正式检验了该基因座的功能边界包含在这段连续基因组范围内的假设。我们发现,两条携带多拷贝413千碱基对连锁Gata2 BAC转基因(携带该基因座从-187至+226千碱基对的序列)的转基因小鼠独立品系能够完全挽救Gata2基因敲除突变体的胚胎致死性,且获救动物的行为和繁殖正常。令人惊讶的是,连锁的BAC赋予输尿管上皮表达,而最初测试的任何重叠亲本BAC和酵母人工染色体中的序列都没有这种表达,因此这些实验还定义了一种以前未描述过的新型合成增强子活性。这些遗传互补研究确定了Gata2基因座所需的外部界限,并正式证明位于这些边界之外的增强子对于Gata2调控细胞活力或繁殖力并非必需。