Literature DB >> 30036593

Genotype-phenotype correlation of patients with tuberous sclerosis complex-associated renal angiomyolipoma: a descriptive study.

Shuqiang Li1, Yushi Zhang2, Zhiyong Wang3, Yanfeng Yang3, Wansheng Gao3, Dongsheng Li3, Jinxing Wei3.   

Abstract

TSC2 gene mutation was repeatedly reported to be associated with a more severe phenotype in patients with tuberous sclerosis complex (TSC). Our current study aims to further explore whether there is such a correlation in patients with TSC-associated renal angiomyolipoma (TSC-RAML). TSC1/TSC2 gene mutation was screened by high-throughput sequencing in 25 TSC-RAML patients from 2 medical centers. Clinical data were also carefully collected. Linear regression analysis and Student t-test were conducted by IBM SPSS Statistics Version 21.0 to analyze the genotypic-phenotypic relationship. The results indicated a high level of TSC gene mutation (80%; 20/25) in TSC-RAML patients, with higher frequency of TSC2 mutation (68%; 17/25) than TSC1 mutation (12%; 3/25). Seven novel mutation sites were detected in this study. In general, there were no significant correlations between tumor size and age (r = 0.134, P = .522), hemoglobin (r = 0.255, P = .219), and serum creatinine (r = 0.043, P = .839). Patients with larger tumor size have higher risk of bleeding. Specially, it was higher hemoglobin level in patients with TSC1 mutation than ones with TSC2 mutation and without TSC mutation (P < .05). However, no difference was found in either tumor size or serum creatinine by TSC mutation genes (P > .05). Furthermore, no difference was found in tumor size, hemoglobin, and serum creatinine by TSC mutation types (P > .05). In conclusion, TSC-RAML is TSC2 mutation dominant, with the individual differences varying greatly. No definite genotype-phenotype correlation exists in patients with TSC-RAML, and it needs to be further explored.
Copyright © 2018 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Genotype; High-throughput sequencing; Phenotype; Renal angiomyolipoma; Tuberous sclerosis complex

Mesh:

Substances:

Year:  2018        PMID: 30036593     DOI: 10.1016/j.humpath.2018.07.017

Source DB:  PubMed          Journal:  Hum Pathol        ISSN: 0046-8177            Impact factor:   3.466


  5 in total

1.  Genetic analysis of 18 families with tuberous sclerosis complex.

Authors:  Kaili Yin; Nan Lin; Qiang Lu; Liri Jin; Yan Huang; Xiangqin Zhou; Kaifeng Xu; Qing Liu; Xue Zhang
Journal:  Neurogenetics       Date:  2022-05-21       Impact factor: 3.017

2.  Analysis of renal lesions in Chinese tuberous sclerosis complex patients with different types of TSC gene mutations.

Authors:  Wenda Wang; Yang Zhao; Xu Wang; Zhan Wang; Yi Cai; Hanzhong Li; Yushi Zhang
Journal:  Genet Mol Biol       Date:  2022-05-27       Impact factor: 2.087

3.  Germline mutation of TSC1 or TSC2 gene in Chinese patients with bilateral renal angiomyolipomas and mutation spectrum of Chinese TSC patients.

Authors:  Wang Jiangyi; Guo Gang; Shi Guohai; Ye Dingwei
Journal:  Aging (Albany NY)       Date:  2020-01-12       Impact factor: 5.682

4.  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

Review 5.  Renal phenotypes correlate with genotypes in unrelated individuals with tuberous sclerosis complex in China.

Authors:  Cong Luo; Ye Zhang; Yu-Shi Zhang; Ming-Xin Zhang; Jun Ning; Min-Feng Chen; Yuan Li; Lin Qi; Xiong-Bing Zu; Yang-Le Li; Yi Cai
Journal:  Orphanet J Rare Dis       Date:  2022-07-23       Impact factor: 4.303

  5 in total

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