{"title":"通过全外显子组测序在伊朗痉挛性截瘫患者中发现ALDH18A1和SPG11基因的两个新突变","authors":"Sajad Rafiee Komachali, Zakieh Siahpoosh, Mansoor Salehi","doi":"10.5808/gi.22030","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":36591,"journal":{"name":"Genomics and Informatics","volume":" ","pages":"e30"},"PeriodicalIF":0.0000,"publicationDate":"2022-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9576469/pdf/","citationCount":"0","resultStr":"{\"title\":\"Two novel mutations in ALDH18A1 and SPG11 gene found by whole-exome sequencing in spastic paraplegia disease patients in Iran.\",\"authors\":\"Sajad Rafiee Komachali, Zakieh Siahpoosh, Mansoor Salehi\",\"doi\":\"10.5808/gi.22030\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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. 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引用次数: 0
摘要
遗传性痉挛性截瘫是一种少见的遗传性神经系统疾病,临床表现具有异质性。ALDH18A1(位于10q24.1)基因相关的痉挛性截瘫(SPG9A和SPG9B)是近年来发现的由显性和隐性突变引起的罕见代谢性疾病。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的临床类型,与SPG11位点(位于15q21.1)有关。痉挛性截瘫有不同的症状,如肌肉萎缩、中度MR、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岁的男性,第二个先证者是一名20岁的女性,他们都患有痉挛性截瘫病。使用基于过滤器的方法从患者、其父母和兄弟姐妹的外周血中提取基因组DNA,并进行量化并用于分子分析和测序。测序文库使用Agilent SureSelect Human All ExonV7试剂盒生成,合格文库送入NovaSeq 6000 Illumina测序仪。Sanger测序采用ABI prism 3730测序仪。本文首次报道了2例病例,1例病例含有ALDH18A1的可能致病性NM_002860: c.475C>T: p.R159X突变,2例病例含有SPG11基因的可能致病性NM_001160227.2: c.5454dupA: p.Glu1819Argfs Ter11突变,均经全外显子组测序鉴定并经Sanger测序证实。我们这项研究的目的是确认这两种新的变异是痉挛性截瘫的直接原因。
Two novel mutations in ALDH18A1 and SPG11 gene found by whole-exome sequencing in spastic paraplegia disease patients in Iran.
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.