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一个新的 GREB1L 基因中的同义突变影响了与泌尿生殖系统发育不全相关的 mRNA 剪接。

A novel de novo synonymous variant in GREB1L impacts the mRNA splicing associated with aplasia of the urogenital system.

机构信息

Medical Genetic Institute of Henan Province, Zhengzhou University People's Hospital, Henan Provincial People's Hospital, Zhengzhou, China.

National Health Commission Key Laboratory of Birth Defects Prevention, Henan Key Laboratory of Population Defects Prevention, Zhengzhou, China.

出版信息

Am J Med Genet A. 2024 Dec;194(12):e63823. doi: 10.1002/ajmg.a.63823. Epub 2024 Aug 2.

Abstract

GREB1-like retinoic acid receptor coactivator (GREB1L) gene is associated with autosomal dominant renal hypodysplasia/aplasia 3 (RHDA3) and deafness, autosomal dominant 80 (DFNA80). Among the GREB1L variants reported, most of them are missense or frameshift, while no pathogenic synonymous variants have been recorded. Classical theory paid little attention to synonymous variants and classified it as nonpathogenic; however, recent studies suggest that the variants might be equally important. Here, we report a 7-year-old girl with new symptoms of clitoromegaly, uterovaginal, and ovarian agenesis as well as right kidney missing. A novel de novo GREB1L synonymous variant (NM_001142966: c.4731C>T, p.G1577=) was identified via whole exome sequencing. The variant was predicted to be disease-causing through in silico analysis and was classified as likely pathogenic. Minigene splicing assays confirmed a 6 bp deletion in mutant cDNA comparing with the wild type, leading to two amino acids lost in GREB1L protein. Secondary and tertiary structure modeling showed alterations in protein structure. Our finding reveals a novel GREB1L variant with a new phenotype of urogenital system and is the first to report a pathogenic synonymous variant in GREB1L which affects mRNA splicing, suggesting synonymous variants cannot be ignored in prenatal diagnosis and genetic counseling.

摘要

GREB1 样视黄酸受体辅激活因子(GREB1L)基因与常染色体显性遗传肾发育不全/发育不良 3 型(RHDA3)和耳聋,常染色体显性遗传 80 型(DFNA80)相关。在所报道的 GREB1L 变异体中,大多数为错义或移码变异体,而没有记录到致病性同义变异体。经典理论很少关注同义变异体,并将其归类为非致病性;然而,最近的研究表明,这些变异体可能同样重要。在这里,我们报告了一例 7 岁女孩出现新的阴蒂肥大、阴道子宫和卵巢发育不全以及右侧肾脏缺失的症状。通过全外显子组测序发现了一个新的 GREB1L 同义变异体(NM_001142966:c.4731C>T,p.G1577=),为新生突变。通过计算机分析预测该变异体可能导致疾病,分类为可能致病性。与野生型相比,突变 cDNA 的迷你基因剪接实验证实存在 6bp 的缺失,导致 GREB1L 蛋白中丢失两个氨基酸。二级和三级结构建模显示蛋白结构发生改变。我们的发现揭示了一种新的 GREB1L 变异体,具有新的泌尿生殖系统表型,并且是首次报道影响 mRNA 剪接的 GREB1L 致病性同义变异体,提示在产前诊断和遗传咨询中不能忽视同义变异体。

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