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通过全外显子组测序在伊朗痉挛性截瘫疾病患者中发现ALDH18A1和SPG11基因的两个新突变。

Two novel mutations in ALDH18A1 and SPG11 gene found by whole-exome sequencing in spastic paraplegia disease patients in Iran.

作者信息

Komachali Sajad Rafiee, Siahpoosh Zakieh, Salehi Mansoor

机构信息

Cellular, Molecular and Genetics Research Center, Isfahan University of Medical Sciences, Isfahan, Iran.

Medical Genetics Research Center of Genome, Isfahan University of Medical Sciences, Isfahan, Iran.

出版信息

Genomics Inform. 2022 Sep;20(3):e30. doi: 10.5808/gi.22030. Epub 2022 Sep 30.

Abstract

Hereditary spastic paraplegia is a not common inherited neurological disorder with heterogeneous clinical expressions. ALDH18A1 (located on 10q24.1) gene-related spastic paraplegias (SPG9A and SPG9B) are rare metabolic disorders caused by dominant and recessive mutations that have been found recently. Autosomal recessive hereditary spastic paraplegia is a common and clinical type of familial spastic paraplegia linked to the SPG11 locus (locates on 15q21.1). There are different symptoms of spastic paraplegia, such as muscle atrophy, moderate MR, short stature, balance problem, and lower limb weakness. Our first proband involves a 45 years old man and our second proband involves a 20 years old woman both are affected by spastic paraplegia disease. Genomic DNA was extracted from the peripheral blood of the patients, their parents, and their siblings using a filter-based methodology and quantified and used for molecular analysis and sequencing. Sequencing libraries were generated using Agilent SureSelect Human All ExonV7 kit, and the qualified libraries are fed into NovaSeq 6000 Illumina sequencers. Sanger sequencing was performed by an ABI prism 3730 sequencer. Here, for the first time, we report two cases, the first one which contains likely pathogenic NM_002860: c.475C>T: p.R159X mutation of the ALDH18A1 and the second one has likely pathogenic NM_001160227.2: c.5454dupA: p.Glu1819Argfs Ter11 mutation of the SPG11 gene and also was identified by the whole-exome sequencing and confirmed by Sanger sequencing. Our aim with this study was to confirm that these two novel variants are direct causes of spastic paraplegia.

摘要

遗传性痉挛性截瘫是一种临床表型多样的罕见遗传性神经疾病。ALDH18A1基因(位于10q24.1)相关的痉挛性截瘫(SPG9A和SPG9B)是由显性和隐性突变引起的罕见代谢性疾病,最近才被发现。常染色体隐性遗传性痉挛性截瘫是一种常见的家族性痉挛性截瘫临床类型,与SPG11基因座(位于15q21.1)相关。痉挛性截瘫有不同的症状,如肌肉萎缩、中度智力障碍、身材矮小、平衡问题和下肢无力。我们的第一个先证者是一名45岁男性,第二个先证者是一名20岁女性,两人均患有痉挛性截瘫疾病。使用基于过滤的方法从患者及其父母和兄弟姐妹的外周血中提取基因组DNA,并进行定量,用于分子分析和测序。使用安捷伦SureSelect Human All ExonV7试剂盒生成测序文库,合格的文库被送入Illumina NovaSeq 6000测序仪。通过ABI prism 3730测序仪进行桑格测序。在此,我们首次报告两例病例,第一例含有可能致病的ALDH18A1基因NM_002860: c.475C>T: p.R159X突变,第二例含有可能致病的SPG11基因NM_001160227.2: c.5454dupA: p.Glu1819Argfs Ter11突变,该突变也通过全外显子组测序鉴定并经桑格测序证实。我们这项研究的目的是确认这两个新变体是痉挛性截瘫的直接病因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6a81/9576469/1130854b3119/gi-22030f1.jpg

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