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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...
Autores principales: | , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
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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. |
format | Online Article Text |
id | pubmed-9865588 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
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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