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Whole-Exome Sequencing Identifies Genetic Variants for Severe Adolescent Idiopathic Scoliosis in a Taiwanese Population

Adolescent idiopathic scoliosis (AIS) is a three-dimensional spinal curvature deformity that appears in the adolescent period. In this study, we performed whole-exome sequencing on 11 unrelated Taiwanese patients with a Cobb’s angle greater than 40 degrees. Our results identified more than 200 poten...

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Autores principales: Lin, Min-Rou, Chou, Po-Hsin, Huang, Kuei-Jung, Ting, Jafit, Liu, Chia-Ying, Chou, Wan-Hsuan, Lin, Gan-Hong, Chang, Jan-Gowth, Ikegawa, Shiro, Wang, Shih-Tien, Chang, Wei-Chiao
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9865588/
https://www.ncbi.nlm.nih.gov/pubmed/36675693
http://dx.doi.org/10.3390/jpm13010032
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author Lin, Min-Rou
Chou, Po-Hsin
Huang, Kuei-Jung
Ting, Jafit
Liu, Chia-Ying
Chou, Wan-Hsuan
Lin, Gan-Hong
Chang, Jan-Gowth
Ikegawa, Shiro
Wang, Shih-Tien
Chang, Wei-Chiao
author_facet Lin, Min-Rou
Chou, Po-Hsin
Huang, Kuei-Jung
Ting, Jafit
Liu, Chia-Ying
Chou, Wan-Hsuan
Lin, Gan-Hong
Chang, Jan-Gowth
Ikegawa, Shiro
Wang, Shih-Tien
Chang, Wei-Chiao
author_sort Lin, Min-Rou
collection PubMed
description Adolescent idiopathic scoliosis (AIS) is a three-dimensional spinal curvature deformity that appears in the adolescent period. In this study, we performed whole-exome sequencing on 11 unrelated Taiwanese patients with a Cobb’s angle greater than 40 degrees. Our results identified more than 200 potential pathogenic rare variants, however, most of which were carried only by one individual. By in silico pathogenicity annotation studies, we found that TTN, CLCN1, and SOX8 were the most important genes, as multiple pathogenic variants were within these genes. Furthermore, biological functional annotation indicated critical roles of these AIS candidate genes in the skeletal muscle. Importantly, a pathogenic variant on SOX8 was shared by over 35% of the patients. These results highlighted TTN, CLCN1, and SOX8 as the most likely susceptibility genes for severe AIS.
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spelling pubmed-98655882023-01-22 Whole-Exome Sequencing Identifies Genetic Variants for Severe Adolescent Idiopathic Scoliosis in a Taiwanese Population Lin, Min-Rou Chou, Po-Hsin Huang, Kuei-Jung Ting, Jafit Liu, Chia-Ying Chou, Wan-Hsuan Lin, Gan-Hong Chang, Jan-Gowth Ikegawa, Shiro Wang, Shih-Tien Chang, Wei-Chiao J Pers Med Article Adolescent idiopathic scoliosis (AIS) is a three-dimensional spinal curvature deformity that appears in the adolescent period. In this study, we performed whole-exome sequencing on 11 unrelated Taiwanese patients with a Cobb’s angle greater than 40 degrees. Our results identified more than 200 potential pathogenic rare variants, however, most of which were carried only by one individual. By in silico pathogenicity annotation studies, we found that TTN, CLCN1, and SOX8 were the most important genes, as multiple pathogenic variants were within these genes. Furthermore, biological functional annotation indicated critical roles of these AIS candidate genes in the skeletal muscle. Importantly, a pathogenic variant on SOX8 was shared by over 35% of the patients. These results highlighted TTN, CLCN1, and SOX8 as the most likely susceptibility genes for severe AIS. MDPI 2022-12-23 /pmc/articles/PMC9865588/ /pubmed/36675693 http://dx.doi.org/10.3390/jpm13010032 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Lin, Min-Rou
Chou, Po-Hsin
Huang, Kuei-Jung
Ting, Jafit
Liu, Chia-Ying
Chou, Wan-Hsuan
Lin, Gan-Hong
Chang, Jan-Gowth
Ikegawa, Shiro
Wang, Shih-Tien
Chang, Wei-Chiao
Whole-Exome Sequencing Identifies Genetic Variants for Severe Adolescent Idiopathic Scoliosis in a Taiwanese Population
title Whole-Exome Sequencing Identifies Genetic Variants for Severe Adolescent Idiopathic Scoliosis in a Taiwanese Population
title_full Whole-Exome Sequencing Identifies Genetic Variants for Severe Adolescent Idiopathic Scoliosis in a Taiwanese Population
title_fullStr Whole-Exome Sequencing Identifies Genetic Variants for Severe Adolescent Idiopathic Scoliosis in a Taiwanese Population
title_full_unstemmed Whole-Exome Sequencing Identifies Genetic Variants for Severe Adolescent Idiopathic Scoliosis in a Taiwanese Population
title_short Whole-Exome Sequencing Identifies Genetic Variants for Severe Adolescent Idiopathic Scoliosis in a Taiwanese Population
title_sort whole-exome sequencing identifies genetic variants for severe adolescent idiopathic scoliosis in a taiwanese population
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9865588/
https://www.ncbi.nlm.nih.gov/pubmed/36675693
http://dx.doi.org/10.3390/jpm13010032
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