Literature DB >> 32917966

Mutation landscape of TSC1/TSC2 in Chinese patients with tuberous sclerosis complex.

Yuhuan Meng1,2, Changshun Yu3, Meijun Chen2, Xiaokang Yu3, Mingming Sun3, Hui Yan1, Weiwei Zhao4,5, Shihui Yu6,7,8.   

Abstract

Genetic testing of TSC1 and TSC2 is important for the diagnosis of tuberous sclerosis complex (TSC), an autosomal dominant neurocutaneous disease. This study retrospectively reviewed 347 samples from patients with clinically suspected TSC being tested for mutations in TSC1 and TSC2 genes using next-generation sequencing and multiplex ligation-dependent probe amplification. Two hundred eighty-one patients (80.98%) were classified as definite/possible/uncertain diagnosis of TSC and the mutational spectrum of TSC1/TSC2 was described. Two hundred eighteen unique nonsynonymous SNVs/Indels (64 in TSC1, 154 in TSC2) and 13 copy number variants (CNVs) were identified in 241 samples (85.77%), including 82 novel variants. CNVs involving 12 large deletions and one duplication were detected exclusively in TSC2. Both TSC1 and TSC2 mutations were nearly uniformly distributed in their protein-coding regions. Furthermore, a string of non-TSC1/TSC2 deleterious variants in 12 genes was identified in the patients, especially overwhelmingly present in the patients with no mutation identified (NMI) in TSC1/TSC2. Our study provides a comprehensive TSC1/TSC2 mutation landscape and reveal some potential risk non-TSCs variants present in patients with NMI.

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Year:  2020        PMID: 32917966     DOI: 10.1038/s10038-020-00839-0

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  2 in total

1.  Genotype-phenotype correlation of renal lesions in the tuberous sclerosis complex.

Authors:  Hitomi Sasaki; Makoto Sumitomo; Yoshinari Muto; Hidehito Inagaki; Maki Kato; Takema Kato; Shunsuke Miyai; Hiroki Kurahashi; Ryoichi Shiroki
Journal:  Hum Genome Var       Date:  2022-02-10

2.  Whole Exome Sequencing in a Series of Patients with a Clinical Diagnosis of Tuberous Sclerosis Not Confirmed by Targeted TSC1/TSC2 Sequencing.

Authors:  Erzsebet Kovesdi; Reka Ripszam; Etelka Postyeni; Emese Beatrix Horvath; Anna Kelemen; Beata Fabos; Viktor Farkas; Kinga Hadzsiev; Katalin Sumegi; Lili Magyari; Pilar Guatibonza Moreno; Peter Bauer; Bela Melegh
Journal:  Genes (Basel)       Date:  2021-09-10       Impact factor: 4.096

  2 in total

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