Gaensler Karin M L, Zhang Zemin, Lin Chin, Yang Suya, Hardt Karin, Flebbe-Rehwaldt Linda
Department of Medicine and Howard Hughes Medical Institute, University of California, San Francisco, CA 94143, USA.
Proc Natl Acad Sci U S A. 2003 Mar 18;100(6):3374-9. doi: 10.1073/pnas.0634132100. Epub 2003 Mar 10.
The human beta-globin locus has been extensively studied as a model of tissue and developmental stage-specific gene expression. Structural mapping of naturally occurring mutations, including transfection and transgenic studies, and the recent finding of intergenic transcripts have suggested that there are cis-acting sequence elements in the (A)gamma-delta intergenic region involved in regulating gamma- and beta-globin gene expression. To determine whether previously identified sequences in the (A)gamma-delta intergenic region are required for appropriate developmental expression of the human beta-globin gene cluster, transgenic mice were generated by transfer of yeast artificial chromosomes containing the entire human beta-globin locus. Three different deletions of the (A)gamma-delta intergenic region were introduced, including (i) deletion of the 750-bp (A)gamma 3' regulatory element ((A)gammae), (ii) deletion of 3.2 kb upstream of the delta-globin gene encompassing pyrimidine-rich sequences and the recently described intergenic transcript initiation site, and (iii) deletion of a 12.5-kb fragment encompassing most of the (A)gamma-delta globin intergenic region. Analysis of multiple transgenic lines carrying these deletion constructs demonstrated that the normal stage-specific sequential expression of the epsilon -, gamma-, and beta-globin genes was preserved, despite deletion of these putative regulatory sequences. These studies suggest that regulatory sequences required for activation and silencing of the human beta-globin gene family during ontogeny reside proximally to the genes and immediately 5' to the human gamma- and beta-globin genes.
人类β-珠蛋白基因座作为组织和发育阶段特异性基因表达的模型已被广泛研究。对自然发生突变的结构定位,包括转染和转基因研究,以及最近对基因间转录本的发现表明,在(A)γ-δ基因间区域存在顺式作用序列元件,参与调节γ-和β-珠蛋白基因的表达。为了确定(A)γ-δ基因间区域中先前鉴定的序列对于人类β-珠蛋白基因簇的适当发育表达是否必需,通过转移包含整个人类β-珠蛋白基因座的酵母人工染色体来生成转基因小鼠。引入了(A)γ-δ基因间区域的三种不同缺失,包括(i)缺失750bp的(A)γ 3'调节元件((A)γe),(ii)缺失δ-珠蛋白基因上游3.2kb包含富含嘧啶序列和最近描述的基因间转录起始位点的区域,以及(iii)缺失包含大部分(A)γ-δ珠蛋白基因间区域的12.5kb片段。对携带这些缺失构建体的多个转基因系的分析表明,尽管缺失了这些假定的调节序列,ε-、γ-和β-珠蛋白基因的正常阶段特异性顺序表达仍得以保留。这些研究表明,在个体发育过程中激活和沉默人类β-珠蛋白基因家族所需的调节序列位于基因近端以及人类γ-和β-珠蛋白基因紧邻的5'端。