Cargando…
Molecular characterization and copy number of SMN1, SMN2 and NAIP in Chinese patients with spinal muscular atrophy and unrelated healthy controls
BACKGROUND: Spinal muscular atrophy (SMA) is caused by SMN1 dysfunction, and the copy number of SMN2 and NAIP can modify the phenotype of SMA. The aim of this study was to analyze the copy numbers and gene structures of SMA-related genes in Chinese SMA patients and unrelated healthy controls. METHOD...
Autores principales: | , , , , , , , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2015
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4328246/ https://www.ncbi.nlm.nih.gov/pubmed/25888055 http://dx.doi.org/10.1186/s12891-015-0457-x |
_version_ | 1782357215523373056 |
---|---|
author | Fang, Ping Li, Liang Zeng, Jian Zhou, Wan-Jun Wu, Wei-Qing Zhong, Ze-Yan Yan, Ti-Zhen Xie, Jian-Sheng Huang, Jing Lin, Li Zhao, Ying Xu, Xiang-Min |
author_facet | Fang, Ping Li, Liang Zeng, Jian Zhou, Wan-Jun Wu, Wei-Qing Zhong, Ze-Yan Yan, Ti-Zhen Xie, Jian-Sheng Huang, Jing Lin, Li Zhao, Ying Xu, Xiang-Min |
author_sort | Fang, Ping |
collection | PubMed |
description | BACKGROUND: Spinal muscular atrophy (SMA) is caused by SMN1 dysfunction, and the copy number of SMN2 and NAIP can modify the phenotype of SMA. The aim of this study was to analyze the copy numbers and gene structures of SMA-related genes in Chinese SMA patients and unrelated healthy controls. METHODS: Forty-two Chinese SMA patients and two hundred and twelve unrelated healthy Chinese individuals were enrolled in our study. The copy numbers and gene structures of SMA-related genes were measured by MLPA assay. RESULTS: We identified a homozygous deletion of SMN1 in exons 7 and 8 in 37 of 42 patients (88.1%); the other 5 SMA patients (11.9%) had a single copy of SMN1 exon 8. The proportions of the 212 unrelated healthy controls with different copy numbers for the normal SMN1 gene were 1 copy in 4 individuals (1.9%), 2 copies in 203 (95.7%) and 3 copies in 5 (2.4%). Three hybrid SMN genes and five genes that lack partial sequences were found in SMA patients and healthy controls. Distributions of copy numbers for normal SMN2 and NAIP were significantly different (P < 0.001) in people with and without SMA. CONCLUSION: The copy numbers and gene structures of SMA-related genes were different in Chinese SMA patients and healthy controls. |
format | Online Article Text |
id | pubmed-4328246 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2015 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-43282462015-02-15 Molecular characterization and copy number of SMN1, SMN2 and NAIP in Chinese patients with spinal muscular atrophy and unrelated healthy controls Fang, Ping Li, Liang Zeng, Jian Zhou, Wan-Jun Wu, Wei-Qing Zhong, Ze-Yan Yan, Ti-Zhen Xie, Jian-Sheng Huang, Jing Lin, Li Zhao, Ying Xu, Xiang-Min BMC Musculoskelet Disord Research Article BACKGROUND: Spinal muscular atrophy (SMA) is caused by SMN1 dysfunction, and the copy number of SMN2 and NAIP can modify the phenotype of SMA. The aim of this study was to analyze the copy numbers and gene structures of SMA-related genes in Chinese SMA patients and unrelated healthy controls. METHODS: Forty-two Chinese SMA patients and two hundred and twelve unrelated healthy Chinese individuals were enrolled in our study. The copy numbers and gene structures of SMA-related genes were measured by MLPA assay. RESULTS: We identified a homozygous deletion of SMN1 in exons 7 and 8 in 37 of 42 patients (88.1%); the other 5 SMA patients (11.9%) had a single copy of SMN1 exon 8. The proportions of the 212 unrelated healthy controls with different copy numbers for the normal SMN1 gene were 1 copy in 4 individuals (1.9%), 2 copies in 203 (95.7%) and 3 copies in 5 (2.4%). Three hybrid SMN genes and five genes that lack partial sequences were found in SMA patients and healthy controls. Distributions of copy numbers for normal SMN2 and NAIP were significantly different (P < 0.001) in people with and without SMA. CONCLUSION: The copy numbers and gene structures of SMA-related genes were different in Chinese SMA patients and healthy controls. BioMed Central 2015-02-07 /pmc/articles/PMC4328246/ /pubmed/25888055 http://dx.doi.org/10.1186/s12891-015-0457-x Text en © Fang et al.; licensee BioMed Central. 2015 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. |
spellingShingle | Research Article Fang, Ping Li, Liang Zeng, Jian Zhou, Wan-Jun Wu, Wei-Qing Zhong, Ze-Yan Yan, Ti-Zhen Xie, Jian-Sheng Huang, Jing Lin, Li Zhao, Ying Xu, Xiang-Min Molecular characterization and copy number of SMN1, SMN2 and NAIP in Chinese patients with spinal muscular atrophy and unrelated healthy controls |
title | Molecular characterization and copy number of SMN1, SMN2 and NAIP in Chinese patients with spinal muscular atrophy and unrelated healthy controls |
title_full | Molecular characterization and copy number of SMN1, SMN2 and NAIP in Chinese patients with spinal muscular atrophy and unrelated healthy controls |
title_fullStr | Molecular characterization and copy number of SMN1, SMN2 and NAIP in Chinese patients with spinal muscular atrophy and unrelated healthy controls |
title_full_unstemmed | Molecular characterization and copy number of SMN1, SMN2 and NAIP in Chinese patients with spinal muscular atrophy and unrelated healthy controls |
title_short | Molecular characterization and copy number of SMN1, SMN2 and NAIP in Chinese patients with spinal muscular atrophy and unrelated healthy controls |
title_sort | molecular characterization and copy number of smn1, smn2 and naip in chinese patients with spinal muscular atrophy and unrelated healthy controls |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4328246/ https://www.ncbi.nlm.nih.gov/pubmed/25888055 http://dx.doi.org/10.1186/s12891-015-0457-x |
work_keys_str_mv | AT fangping molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT liliang molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT zengjian molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT zhouwanjun molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT wuweiqing molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT zhongzeyan molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT yantizhen molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT xiejiansheng molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT huangjing molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT linli molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT zhaoying molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols AT xuxiangmin molecularcharacterizationandcopynumberofsmn1smn2andnaipinchinesepatientswithspinalmuscularatrophyandunrelatedhealthycontrols |