结节性硬化症TSC1和TSC2基因分析及表型相关性评价。

IF 2.3 3区 生物学 Q3 BIOCHEMISTRY & MOLECULAR BIOLOGY
Metin Eser, Gulam Hekimoglu, Busra Kutlubay, Safiye Gunes Sager, Ayberk Turkyilmaz
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引用次数: 0

摘要

结节性硬化症(TSC)是一种罕见的遗传性疾病,其特征是在各器官,特别是中枢神经系统形成良性肿瘤。我们的目的是通过使用下一代测序(NGS)分析TSC1和TSC2基因来描绘被诊断为TSC的土耳其个体的分子谱。Sophia Genetics的Sophia遗传病小组对22名被诊断为TSC的个体进行了NGS,并鉴定了TSC1和TSC2基因的致病变异。22例中,TSC1突变3例(13.6%),TSC2突变16例(73%),3例(13.6%)未检出突变。值得注意的是,一名TSC2突变患者表现为血管纤维瘤、足趾纤维瘤和牙釉质凹陷,而另一名患者表现为心脏横纹肌瘤。6例(27%)TSC2突变患者出现自闭症谱系障碍,其中1例有自闭症行为。运动发育异常3例(13.6%),其中2例有TSC2突变。TSC2突变3例(13.6%)存在严重智力残疾,TSC2突变2例(9%)存在发育迟缓。3例(13.6%)发生癫痫性脑病,2例发生TSC2突变。此外,6例(27%)表现出局灶性癫痫耐药,其中5例有TSC2突变。这些发现与其他研究一致,表明与TSC1突变相比,TSC2突变与更严重的表型范围相关。此外,我们的分析显示,一些TSC1/TSC2突变的人不符合诊断标准。这突出了基因检测和分子谱分析在理解临床变异性和帮助TSC患者管理方面的重要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Analysis of TSC1 and TSC2 genes and evaluation of phenotypic correlations with tuberous sclerosis.

Tuberous sclerosis complex (TSC) is a rare genetic disorder characterized by the formation of benign tumors in various organs, particularly in the central nervous system. We aimed to delineate the molecular profile of Turkish individuals diagnosed with TSC by analyzing the TSC1 and TSC2 genes using next-generation sequencing (NGS). Sophia Genetics' Sophia Inherited Disease Panel was used to perform NGS on 22 individuals diagnosed with TSC and to identify pathogenic variants in the TSC1 and TSC2 genes. Among the 22 cases, mutations were found in 3 (13.6%) for TSC1 and in 16 (73%) for TSC2, while 3 (13.6%) exhibited no detectable mutations. Notably, one individual with a TSC2 mutation presented with angiofibroma, ungual fibroma, and pitted dental enamel, while another had cardiac rhabdomyoma. Autism spectrum disorders were observed in 6 (27%) with TSC2 mutations, including one with autistic behavior. Abnormal motor development was noted in 3 (13.6%), of which 2 had TSC2 mutations. Severe intellectual disability was found in 3 (13.6%) with TSC2 mutations, and developmental delay was seen in 2 (9%) with TSC2 mutations. Epileptic encephalopathy occurred in 3 (13.6%), with 2 having TSC2 mutations. Additionally, 6 (27%) exhibited drug resistance for focal seizures, with 5 of them having TSC2 mutations. These findings are consistent with other research indicating that TSC2 mutations are associated with a more severe phenotypic range compared to TSC1 mutations. Moreover, our analysis showed that some people with TSC1/TSC2 mutations did not match diagnostic criteria. This highlights the importance of genetic testing and molecular profiling in understanding the clinical variability and aiding in the management of TSC patients.

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来源期刊
Molecular Genetics and Genomics
Molecular Genetics and Genomics 生物-生化与分子生物学
CiteScore
5.10
自引率
3.20%
发文量
134
审稿时长
1 months
期刊介绍: Molecular Genetics and Genomics (MGG) publishes peer-reviewed articles covering all areas of genetics and genomics. Any approach to the study of genes and genomes is considered, be it experimental, theoretical or synthetic. MGG publishes research on all organisms that is of broad interest to those working in the fields of genetics, genomics, biology, medicine and biotechnology. The journal investigates a broad range of topics, including these from recent issues: mechanisms for extending longevity in a variety of organisms; screening of yeast metal homeostasis genes involved in mitochondrial functions; molecular mapping of cultivar-specific avirulence genes in the rice blast fungus and more.
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