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韩国1型神经纤维瘤病患者中NF1剪接突变的鉴定与特征分析

Identification and characterization of NF1 splicing mutations in Korean patients with neurofibromatosis type 1.

作者信息

Jang Mi-Ae, Kim Young-Eun, Kim Sun Kyung, Lee Myoung-Keun, Kim Jong-Won, Ki Chang-Seok

机构信息

Department of Laboratory Medicine and Genetics, Soonchunhyang University Bucheon Hospital, Soonchunhyang University College of Medicine, Bucheon, Republic of Korea.

Green Cross Genome, Yongin, Republic of Korea.

出版信息

J Hum Genet. 2016 Aug;61(8):705-9. doi: 10.1038/jhg.2016.33. Epub 2016 Apr 14.

DOI:10.1038/jhg.2016.33
PMID:27074763
Abstract

Neurofibromatosis type I (NF1) is an autosomal dominant genetic disorder caused by NF1 mutations. Although mutations affecting mRNA splicing are the most common molecular defects in NF1, few studies have analyzed genomic DNA (gDNA)-mRNA correlations in Korean NF1 patients. In this study, we investigated 28 unrelated NF1 patients who showed splicing alterations in reverse transcription-PCR of NF1 mRNA and identified 24 different NF1 splicing mutations, 9 of which were novel. These mutations can be categorized into five groups: exon skipping resulting from mutations at authentic 5' and 3' splice sites (type I, 46%), cryptic exon inclusion caused by deep intronic mutations (type II, 8%), creation of new splice sites causing loss of exonic sequences (type III, 8%), activation of cryptic splice sites due to disruption of authentic splice sites (type IV, 25%) and exonic sequence alterations causing exon skipping (type V, 13%). In total, 42% of all splicing mutations did not involve the conserved AG/GT dinucleotides of the splice sites, making it difficult to identify the correct mutation sites at the gDNA level. These results add to the mutational spectrum of NF1 and further elucidate the gDNA-mRNA correlations of NF1 mutations.

摘要

I型神经纤维瘤病(NF1)是一种由NF1基因突变引起的常染色体显性遗传病。尽管影响mRNA剪接的突变是NF1中最常见的分子缺陷,但很少有研究分析韩国NF1患者的基因组DNA(gDNA)与mRNA的相关性。在本研究中,我们调查了28例无亲缘关系的NF1患者,这些患者在NF1 mRNA的逆转录PCR中显示出剪接改变,并鉴定出24种不同的NF1剪接突变,其中9种是新的。这些突变可分为五组:由真实5'和3'剪接位点突变导致的外显子跳跃(I型,46%)、由内含子深处突变引起的隐蔽外显子包含(II型,8%)、导致外显子序列丢失的新剪接位点的产生(III型,8%)、由于真实剪接位点破坏导致的隐蔽剪接位点激活(IV型,25%)以及导致外显子跳跃的外显子序列改变(V型,13%)。总体而言,所有剪接突变中有42%不涉及剪接位点保守的AG/GT二核苷酸,这使得在gDNA水平上难以确定正确的突变位点。这些结果增加了NF1的突变谱,并进一步阐明了NF1突变的gDNA与mRNA的相关性。

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Genotype-Phenotype Correlations in Neurofibromatosis Type 1: Identification of Novel and Recurrent Gene Variants and Correlations with Neurocognitive Phenotype.神经纤维瘤病 1 型的基因型-表型相关性:新的和反复出现的基因突变的鉴定及其与神经认知表型的相关性。
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Front Genet. 2021 Nov 19;12:772958. doi: 10.3389/fgene.2021.772958. eCollection 2021.
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