Analysis of TSC1 and TSC2 genes and evaluation of phenotypic correlations with tuberous sclerosis

被引:0
|
作者
Eser, Metin [1 ]
Hekimoglu, Gulam [2 ]
Kutlubay, Busra [3 ]
Sager, Safiye Gunes [4 ]
Turkyilmaz, Ayberk [5 ]
机构
[1] Univ Hlth Sci, Umraniye Educ & Res Hosp, Dept Med Genet, Istanbul, Turkiye
[2] Univ Hlth Sci, Hamidiye Int Fac Med, Dept Histol & Embryol, Istanbul, Turkiye
[3] Univ Hlth Sci, Umraniye Educ & Res Hosp, Dept Pediat Neurol, Istanbul, Turkiye
[4] Univ Hlth Sci, Kartal Dr Lutfi Kirdar City Hosp, Dept Pediat Neurol, Istanbul, Turkiye
[5] Karadeniz Tech Univ, Fac Med, Dept Med Genet, Trabzon, Turkiye
关键词
TSC1; TSC2; Tuberous Sclerosis; Autosomal dominant; NGS; COMPLEX; SURVEILLANCE; INDIVIDUALS; DISORDERS; DIAGNOSIS;
D O I
10.1007/s00438-024-02210-w
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
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.
引用
收藏
页数:10
相关论文
共 50 条
  • [1] Molecular analysis of TSC1 and TSC2 genes and phenotypic correlations in Brazilian families with tuberous sclerosis
    Rosset, Clevia
    Vairo, Filippo
    Bandeira, Isabel Cristina
    Correia, Rudinei Luis
    de Goes, Fernanda Veiga
    Boy da Silva, Raquel Tavares
    Mario Bueno, Larissa Souza
    Silva de Miranda Gomes, Mireille Caroline
    Reis Galvao, Henrique de Campos
    Neri, Joao I. C. F.
    Achatz, Maria Isabel
    Oliveira Netto, Cristina Brinckmann
    Ashton-Prolla, Patricia
    PLOS ONE, 2017, 12 (10):
  • [2] Comprehensive mutation analysis of TSC1 and TSC2 -: and phenotypic correlations in 150 families with tuberous sclerosis
    Jones, AC
    Shyamsundar, MM
    Thomas, MW
    Maynard, J
    Idziaszczyk, S
    Tomkins, S
    Sampson, JR
    Cheadle, JP
    AMERICAN JOURNAL OF HUMAN GENETICS, 1999, 64 (05) : 1305 - 1315
  • [3] Tuberous Sclerosis Complex in Chinese patients: Phenotypic analysis and mutational screening of TSC1/TSC2 genes
    Lin, Shan
    Zeng, Jia-Bin
    Zhao, Gui-Xian
    Yang, Zhen-Zhen
    Huang, Hui-Ping
    Lin, Min-Ting
    Wu, Zhi-Ying
    Wang, Ning
    Chen, Wan-Jin
    Fang, Ling
    SEIZURE-EUROPEAN JOURNAL OF EPILEPSY, 2019, 71 : 322 - 327
  • [4] Mutational analysis of TSC1 and TSC2 genes in Japanese patients with tuberous sclerosis complex
    Zhang, H
    Nanba, E
    Yamamoto, T
    Ninomiya, H
    Ohno, K
    Mizuguchi, M
    Takeshita, K
    JOURNAL OF HUMAN GENETICS, 1999, 44 (06) : 391 - 396
  • [5] Mutational analysis of TSC1 and TSC2 genes in Japanese patients with tuberous sclerosis complex
    H. Zhang
    E. Nanba
    T. Yamamoto
    H. Ninomiya
    K. Ohno
    M. Mizuguchi
    K. Takeshita
    Journal of Human Genetics, 1999, 44 : 391 - 396
  • [6] TSC1 and TSC2: genes that are mutated in the human genetic disorder tuberous sclerosis
    Sampson, JR
    BIOCHEMICAL SOCIETY TRANSACTIONS, 2003, 31 : 592 - 596
  • [7] Mutational analysis of TSC1 and TSC2 genes in Tuberous Sclerosis Complex patients from Greece
    Socratis Avgeris
    Florentia Fostira
    Andromachi Vagena
    Yiannis Ninios
    Angeliki Delimitsou
    Radek Vodicka
    Radek Vrtel
    Sotirios Youroukos
    Dimitrios J. Stravopodis
    Metaxia Vlassi
    Aristotelis Astrinidis
    Drakoulis Yannoukakos
    Gerassimos E. Voutsinas
    Scientific Reports, 7
  • [8] Mutation and polymorphism analysis of TSC1 and TSC2 genes in Indian patients with tuberous sclerosis complex
    Ali, M
    Girimaji, SC
    Markandaya, M
    Shukla, AK
    Sacchidanand, S
    Kumar, A
    ACTA NEUROLOGICA SCANDINAVICA, 2005, 111 (01): : 54 - 63
  • [9] Mutational analysis of TSC1 and TSC2 genes in Tuberous Sclerosis Complex patients from Greece
    Avgeris, Socratis
    Fostira, Florentia
    Vagena, Andromachi
    Ninios, Yiannis
    Delimitsou, Angeliki
    Vodicka, Radek
    Vrtel, Radek
    Youroukos, Sotirios
    Stravopodis, Dimitrios J.
    Vlassi, Metaxia
    Astrinidis, Aristotelis
    Yannoukakos, Drakoulis
    Voutsinas, Gerassimos E.
    SCIENTIFIC REPORTS, 2017, 7
  • [10] TSC1 and TSC2 mutations in tuberous sclerosis, the associated phenotypes and a model to explain observed TSC1/TSC2 frequency ratios
    Nicola Langkau
    Nicola Martin
    Regine Brandt
    Karin Zügge
    Stefanie Quast
    Gerd Wiegele
    Anna Jauch
    Marion Rehm
    Andrea Kuhl
    Monika Mack-Vetter
    Lothar Zimmerhackl
    Bart Janssen
    European Journal of Pediatrics, 2002, 161 : 393 - 402