Li Mingjie, Lu Xinxin, Dong Jian, Yao Zuwu, Wu Yinlong, Rao Huiying, Huang Xiaoli, Chen Xijun, Huang Yi, Wu Yan'an
Department of Clinical Laboratory, Fujian Provincial Hospital, Shengli Clinical Medical College, Fujian Medical University, Fuzhou 350001, China.
Department of Cardiovascular Surgery, Fujian Provincial Hospital, Fuzhou 350001, China.
Genomics. 2020 Nov;112(6):3856-3861. doi: 10.1016/j.ygeno.2020.06.024. Epub 2020 Jun 17.
Marfan syndrome is a heritable autosomal-dominant connective tissue disorder and it was typically caused by mutations in FBN1. However, the synonymous mutation was seldom recorded to be related to Marfan syndrome. Hereon, Multiplex ligation-dependent probe amplification failed to detect a copy number variant involving FBN1 but a synonymous mutation c.4773A > G (p.Gly1591Gly) was identified by NGS in exon 39. RNA was extracted from patient's aortic tissue and reverse polymerase chain reaction demonstrated the presence of a shortened mRNA transcript. Results of minigene models indicated that c.4773A > G was bona fide responsibility for the aberrant splicing pattern, and artificial mutations of c.4773A > C and c.4773A > T also gave rise to fragments with exon 39 entire skipped. Together, the novel synonymous mutations in c.4773 position (A > G, C, T), middle of exon 39 of FBN1 gene, was found to be associated with Marfan syndrome by altering the splicing pattern of pre-mRNA.
马凡综合征是一种遗传性常染色体显性结缔组织疾病,通常由FBN1基因突变引起。然而,同义突变很少被记录为与马凡综合征有关。在此,多重连接依赖探针扩增未能检测到涉及FBN1的拷贝数变异,但通过二代测序在外显子39中鉴定出一个同义突变c.4773A>G(p.Gly1591Gly)。从患者主动脉组织中提取RNA,逆转录聚合酶链反应证明存在缩短的mRNA转录本。小基因模型结果表明,c.4773A>G是异常剪接模式的真正原因,c.4773A>C和c.4773A>T的人工突变也导致外显子39完全跳过的片段。总之,发现FBN1基因外显子39中部c.4773位置(A>G、C、T)的新型同义突变通过改变前体mRNA的剪接模式与马凡综合征相关。