Department of Cardiac Surgery, Beijing Anzhen Hospital, Capital Medical University, No.2, Anzhen Road, Chaoyang District, Beijing, 100029, China.
Key Laboratory of Medical Engineering for Cardiovascular Disease, Ministry of Education, Beijing, China.
Hereditas. 2021 Jan 6;158(1):6. doi: 10.1186/s41065-020-00170-w.
Marfan syndrome (MFS) is one of the most common hereditary connective tissue diseases, with great individual heterogeneity. We reported a Chinese pregnancy with Clinical diagnosis of MFS, performed whole-exome sequencing, and screened for the genetic abnormality. We also conducted an in vitro mini-gene splicing assay to demonstrate the predicted harmful effects of an intronic variant of FBN-1. Exome sequencing identified a novel intronic variant (c.6497-13 T>A) in intron 53 of the FBN-1 gene (NM_000138.4). It's predicted to insert 11 bp of intron 53 into the mature mRNA. The mini-gene splicing experiment demonstrated that c.6497-13 T>A could result in 11 bp retention in intron 53 to exon 54 (c.6496_6497ins gtttcttgcag) and the use of an alternative donor causing the frameshift p.Asp2166Glyfs*23. According to the results, the pregnant woman chose to continue the pregnancy and gave birth to a healthy baby. This study expands the genetic mutation spectrum of MFS patients and indicates the importance of intron sequencing.
马凡综合征(MFS)是最常见的遗传性结缔组织疾病之一,具有很大的个体异质性。我们报告了一例临床诊断为 MFS 的中国妊娠病例,进行了全外显子组测序,并筛查了遗传异常。我们还进行了体外迷你基因剪接分析,以证明 FBN-1 内含子变异的预测有害影响。外显子组测序在 FBN-1 基因(NM_000138.4)的内含子 53 中发现了一个新的内含子变异(c.6497-13T>A)。它预测会将 11bp 的内含子 53 插入成熟 mRNA 中。迷你基因剪接实验表明,c.6497-13T>A 可能导致内含子 53 中的 11bp 保留到外显子 54(c.6496_6497ins gtttcttgcag),并使用替代供体导致移码 p.Asp2166Glyfs*23。根据结果,孕妇选择继续妊娠并生下了一个健康的婴儿。这项研究扩展了 MFS 患者的基因突变谱,并表明内含子测序的重要性。