Sorolla Anabel, Tallack Michael R, Oey Harald, Harten Sarah K, Daxinger Lucia Clemens-, Magor Graham W, Combes Alex N, Ilsley Melissa, Whitelaw Emma, Perkins Andrew C
Epigenetics Laboratory, QIMR Berghofer Medical Research Institute, Herston, QLD 4006, Australia.
Mater-UQ Research Institute, The University of Queensland, Translational Research Institute, Woolloongabba, QLD 4102, Australia; School of Medicine, The University of Queensland, St Lucia, QLD 4072, Australia.
Genomics. 2015 Feb;105(2):116-22. doi: 10.1016/j.ygeno.2014.09.013. Epub 2014 Oct 31.
Position-effect variegation of transgene expression is sensitive to the chromatin state. We previously reported a forward genetic screen in mice carrying a variegated α-globin GFP transgene to find novel genes encoding epigenetic regulators. We named the phenovariant strains "Mommes" for modifiers of murine metastable epialleles. Here we report positional cloning of mutations in two Momme strains which result in suppression of variegation. Both strains harbour point mutations in the erythroid transcription factor, Klf1. One (D11) generates a stop codon in the zinc finger domain and a homozygous null phenotype. The other (D45) generates an amino acid transversion (H350R) within a conserved linker between zinc fingers two and three. Homozygous MommeD45 mice have chronic microcytic anaemia which models the phenotype in a recently described family. This is the first genetic evidence that the linkers between the zinc fingers of transcription factors have a function beyond that of a simple spacer.
转基因表达的位置效应斑驳对染色质状态敏感。我们之前报道了在携带斑驳α-珠蛋白绿色荧光蛋白转基因的小鼠中进行的正向遗传筛选,以寻找编码表观遗传调控因子的新基因。我们将表型变异株命名为“Mommes”,即小鼠亚稳定表观等位基因的修饰因子。在此我们报道了两个Momme株系中导致斑驳抑制的突变的定位克隆。两个株系在红系转录因子Klf1中都存在点突变。一个(D11)在锌指结构域产生一个终止密码子,表现为纯合无效表型。另一个(D45)在锌指二和锌指三之间的保守连接区内产生一个氨基酸转换(H350R)。纯合的MommeD45小鼠患有慢性小细胞贫血,这与最近描述的一个家族中的表型相似。这是首个遗传学证据,表明转录因子锌指之间的连接区具有超出简单间隔功能的作用。