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EDA基因突变是中国家系中X连锁少汗性外胚层发育不良和牙发育不全的病因。

Mutations in the EDA gene are responsible for X-linked hypohidrotic ectodermal dysplasia and hypodontia in Chinese kindreds.

作者信息

Fan Huali, Ye Xiaoqian, Shi Lisong, Yin Wei, Hua Bo, Song Guangtai, Shi Bin, Bian Zhuan

机构信息

Key Laboratory for Oral Biomedical Engineering of Ministry of Education, Department of Endodontics, Hospital and School of Stomatology, Wuhan University, Hubei, China.

出版信息

Eur J Oral Sci. 2008 Oct;116(5):412-7. doi: 10.1111/j.1600-0722.2008.00555.x.


DOI:10.1111/j.1600-0722.2008.00555.x
PMID:18821982
Abstract

X-linked hypohidrotic ectodermal dysplasia (XLHED, OMIM 305100) is a rare congenital disorder that results in the defective development of teeth, hair, nails, and eccrine sweat glands. Previous studies found that mutations in the ectodysplasin A (EDA) gene are associated with XLHED. In the present study, we investigated four Chinese families suffering from classical XLHED and investigated two additional families segregating hypodontia in an X-linked recessive manner. Mutations were characterized respectively in the EDA gene in all families, and five of these mutations were found to be novel. Among these mutations, five were missense (c.200A>T, c.463C>T, c.758T>C, c.926T>G, and c.491A>C) and located in the functional domain of EDA, and one was a splice donor site mutation in intron 5 (c.IVS5 + 1G>A), which may result in an alternative transcript derived from a new cryptic splice site. Our data further confirm that EDA mutations could cause both XLHED and isolated hypodontia and provide evidence that EDA is a strong candidate gene for tooth genesis.

摘要

X连锁少汗型外胚层发育不良(XLHED,OMIM 305100)是一种罕见的先天性疾病,会导致牙齿、毛发、指甲和外分泌汗腺发育缺陷。先前的研究发现,外胚层发育不良蛋白A(EDA)基因突变与XLHED有关。在本研究中,我们调查了四个患有典型XLHED的中国家庭,并另外调查了两个以X连锁隐性方式分离牙列缺损的家庭。分别对所有家庭的EDA基因进行了突变特征分析,发现其中五个突变是新的。在这些突变中,五个是错义突变(c.200A>T、c.463C>T c.758T>C、c.926T>G和c.491A>C),位于EDA的功能域,一个是内含子5的剪接供体位点突变(c.IVS5+1G>A),这可能导致来自新的隐蔽剪接位点的替代转录本。我们的数据进一步证实,EDA突变可导致XLHED和孤立性牙列缺损,并提供证据表明EDA是牙齿发育的一个强有力的候选基因。

相似文献

[1]
Mutations in the EDA gene are responsible for X-linked hypohidrotic ectodermal dysplasia and hypodontia in Chinese kindreds.

Eur J Oral Sci. 2008-10

[2]
Novel EDA mutation in X-linked hypohidrotic ectodermal dysplasia and genotype-phenotype correlation.

Oral Dis. 2015-11

[3]
A common founder mutation in the EDA-A1 gene in X-linked hypodontia.

Dermatology. 2010

[4]
Mutation p.Leu354Pro in EDA causes severe hypohidrotic ectodermal dysplasia in a Chinese family.

Gene. 2011-10-10

[5]
Mutation Screening of the EDA Gene in Seven Chinese Families with X-Linked Hypohidrotic Ectodermal Dysplasia.

Genet Test Mol Biomarkers. 2018-8

[6]
Correlation between the phenotypes and genotypes of X-linked hypohidrotic ectodermal dysplasia and non-syndromic hypodontia caused by ectodysplasin-A mutations.

Eur J Med Genet. 2011

[7]
A novel de novo frame-shift mutation of the EDA gene in a Chinese Han family with hypohidrotic ectodermal dysplasia.

J Hum Genet. 2006

[8]
A novel missense mutation p.S305R of EDA gene causes XLHED in a Chinese family.

Arch Oral Biol. 2019-7-24

[9]
Methylation state of the EDA gene promoter in Chinese X-linked hypohidrotic ectodermal dysplasia carriers.

PLoS One. 2013-4-23

[10]
A novel frameshift mutation of the EDA1 gene in a Chinese Han family with X-linked hypohidrotic ectodermal dysplasia.

Clin Exp Dermatol. 2009-1

引用本文的文献

[1]
Mutations Causing X-Linked Recessive Oligodontia with Variable Expression.

Genes (Basel). 2024-12-26

[2]
Genotypic and phenotypic correlations in tooth agenesis: insights from WNT10A and EDA mutations in syndromic and non-syndromic forms.

Hum Genet. 2024-11

[3]
The EDA/EDAR/NF-κB pathway in non-syndromic tooth agenesis: A genetic perspective.

Front Genet. 2023-4-3

[4]
A novel mutation in the collagen domain of EDA results in hypohidrotic ectodermal dysplasia by impacting the receptor-binding capability.

Mol Genet Genomic Med. 2023-4

[5]
Rethinking the Genetic Etiology of Nonsyndromic Tooth Agenesis.

Curr Osteoporos Rep. 2022-12

[6]
Two novel mutations in MSX1 causing oligodontia.

PLoS One. 2020-1-8

[7]
A novel frameshift mutation in the gene in an Iranian patient affected by X-linked hypohidrotic ectodermal dysplasia.

Cell Mol Biol Lett. 2019-8-19

[8]
Novel EDA or EDAR Mutations Identified in Patients with X-Linked Hypohidrotic Ectodermal Dysplasia or Non-Syndromic Tooth Agenesis.

Genes (Basel). 2017-10-5

[9]
Genetic Basis of Nonsyndromic and Syndromic Tooth Agenesis.

J Pediatr Genet. 2016-12

[10]
Methylation state of the EDA gene promoter in Chinese X-linked hypohidrotic ectodermal dysplasia carriers.

PLoS One. 2013-4-23

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