Koller Ulrich, Hainzl Stefan, Kocher Thomas, Hüttner Clemens, Klausegger Alfred, Gruber Christina, Mayr Elisabeth, Wally Verena, Bauer Johann W, Murauer Eva M
Department of Dermatology and EB House Austria, Paracelsus Medical University, Salzburg 5020, Austria.
Int J Mol Sci. 2015 Jan 6;16(1):1179-91. doi: 10.3390/ijms16011179.
Spliceosome-mediated RNA trans-splicing has become an emergent tool for the repair of mutated pre-mRNAs in the treatment of genetic diseases. RNA trans-splicing molecules (RTMs) are designed to induce a specific trans-splicing reaction via a binding domain for a respective target pre-mRNA region. A previously established reporter-based screening system allows us to analyze the impact of various factors on the RTM trans-splicing efficiency in vitro. Using this system, we are further able to investigate the potential of antisense RNAs (AS RNAs), presuming to improve the trans-splicing efficiency of a selected RTM, specific for intron 102 of COL7A1. Mutations in the COL7A1 gene underlie the dystrophic subtype of the skin blistering disease epidermolysis bullosa (DEB). We have shown that co-transfections of the RTM and a selected AS RNA, interfering with competitive splicing elements on a COL7A1-minigene (COL7A1-MG), lead to a significant increase of the RNA trans-splicing efficiency. Thereby, accurate trans-splicing between the RTM and the COL7A1-MG is represented by the restoration of full-length green fluorescent protein GFP on mRNA and protein level. This mechanism can be crucial for the improvement of an RTM-mediated correction, especially in cases where a high trans-splicing efficiency is required.
剪接体介导的RNA反式剪接已成为治疗遗传疾病中修复突变前体mRNA的一种新兴工具。RNA反式剪接分子(RTM)被设计用于通过与各自目标前体mRNA区域的结合域诱导特定的反式剪接反应。一个先前建立的基于报告基因的筛选系统使我们能够在体外分析各种因素对RTM反式剪接效率的影响。利用该系统,我们进一步能够研究反义RNA(AS RNA)的潜力,推测其可提高针对COL7A1第102内含子的选定RTM的反式剪接效率。COL7A1基因的突变是皮肤水疱病大疱性表皮松解症(DEB)营养不良亚型的基础。我们已经表明,RTM与选定的AS RNA共转染,干扰COL7A1小基因(COL7A1-MG)上的竞争性剪接元件,会导致RNA反式剪接效率显著提高。因此,RTM与COL7A1-MG之间准确的反式剪接表现为全长绿色荧光蛋白GFP在mRNA和蛋白质水平上的恢复。这种机制对于改善RTM介导的校正可能至关重要,特别是在需要高反式剪接效率的情况下。