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对来自希腊的结节性硬化症患者 TSC1 和 TSC2 基因的突变分析。

Mutational analysis of TSC1 and TSC2 genes in Tuberous Sclerosis Complex patients from Greece.

机构信息

Laboratory of Environmental Mutagenesis and Carcinogenesis, Institute of Biosciences and Applications, National Centre for Scientific Research "Demokritos", Athens, Greece.

Molecular Diagnostics Laboratory, INRaSTES, National Centre for Scientific Research "Demokritos", Athens, Greece.

出版信息

Sci Rep. 2017 Dec 1;7(1):16697. doi: 10.1038/s41598-017-16988-w.

Abstract

Tuberous sclerosis complex (TSC) is a rare autosomal dominant disorder causing benign tumors in the brain and other vital organs. The genes implicated in disease development are TSC1 and TSC2. Here, we have performed mutational analysis followed by a genotype-phenotype correlation study based on the clinical characteristics of the affected individuals. Twenty unrelated probands or families from Greece have been analyzed, of whom 13 had definite TSC, whereas another 7 had a possible TSC diagnosis. Using direct sequencing, we have identified pathogenic mutations in 13 patients/families (6 in TSC1 and 7 in TSC2), 5 of which were novel. The mutation identification rate for patients with definite TSC was 85%, but only 29% for the ones with a possible TSC diagnosis. Multiplex ligation-dependent probe amplification (MLPA) did not reveal any genomic rearrangements in TSC1 and TSC2 in the samples with no mutations identified. In general, TSC2 disease was more severe than TSC1, with more subependymal giant cell astrocytomas and angiomyolipomas, higher incidence of pharmacoresistant epileptic seizures, and more severe neuropsychiatric disorders. To our knowledge, this is the first comprehensive TSC1 and TSC2 mutational analysis carried out in TSC patients in Greece.

摘要

结节性硬化症复合征(TSC)是一种罕见的常染色体显性遗传疾病,会导致大脑和其他重要器官的良性肿瘤。与疾病发展相关的基因是 TSC1 和 TSC2。在这里,我们根据受影响个体的临床特征进行了突变分析和基因型-表型相关性研究。分析了来自希腊的 20 名无关先证者或家族,其中 13 名患有明确的 TSC,另有 7 名可能患有 TSC。通过直接测序,我们在 13 名患者/家庭(6 名在 TSC1 中,7 名在 TSC2 中)中发现了致病性突变,其中 5 个是新的。明确 TSC 患者的突变识别率为 85%,但可能 TSC 诊断的患者只有 29%。在未发现突变的样本中,多重连接依赖性探针扩增(MLPA)未发现 TSC1 和 TSC2 的基因组重排。一般来说,TSC2 疾病比 TSC1 更严重,有更多的室管膜下巨细胞星形细胞瘤和血管平滑肌脂肪瘤,抗药性癫痫发作的发生率更高,神经精神障碍更严重。据我们所知,这是在希腊进行的首例全面的 TSC1 和 TSC2 突变分析。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/153f/5711901/ecaa9383de70/41598_2017_16988_Fig1_HTML.jpg

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