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一名TP73L基因突变儿童的海-韦综合征

Hay-Wells syndrome in a child with mutation in the TP73L gene.

作者信息

Garcia Bartels Natalie, Neumann Luitgard M, Mleczko Anna, Rubach Katharina, Peters Hartmut, Rossi Rainer, Sterry Wolfram, Blume-Peytavi Ulrike

机构信息

Department of Dermatology and Allergy, Charité- Universitätsmedizin Berlin, Germany.

出版信息

J Dtsch Dermatol Ges. 2007 Oct;5(10):919-23. doi: 10.1111/j.1610-0387.2007.06379.x.


DOI:10.1111/j.1610-0387.2007.06379.x
PMID:17910675
Abstract

Hay-Wells syndrome is a rare form of ectodermal dysplasia, also known as AEC syndrome (Ankyloblepharon filiforme adnatum, Ectodermal effects, Cleft lip/palate). It is inherited in an autosomal dominant fashion with variable expression, featuring congenital abnormalities of skin, hair, teeth, nail, eccrine and mucous glands. We present a three-month-old boy, born to unaffected parents, with typical clinical findings of AEC syndrome. In this boy, a mutation Ile537Thr (c.1610C>T) in the sterile alpha motive (SAM) domain of the TP73L (p63) gene was detected. Because of the broad spectrum of related syndromes such as Rapp-Hodgkin syndrome, Bowen-Armstrong syndrome, CHAND syndrome and epidermolysis bullosa hereditaria, the diagnosis of AEC should be base don both clinical findings and genetic analysis.

摘要

海-韦综合征是一种罕见的外胚层发育不良,也称为AEC综合征(丝状黏连性睑缘粘连、外胚层效应、唇腭裂)。它以常染色体显性方式遗传,表现多样,具有皮肤、毛发、牙齿、指甲、小汗腺和黏液腺的先天性异常。我们报告一名3个月大的男婴,其父母无病,具有典型的AEC综合征临床症状。在这个男孩中,检测到TP73L(p63)基因无菌α基序(SAM)结构域中的Ile537Thr(c.1610C>T)突变。由于相关综合征范围广泛,如拉普-霍奇金综合征、鲍恩-阿姆斯特朗综合征、CHAND综合征和遗传性大疱性表皮松解症,AEC的诊断应基于临床症状和基因分析。

相似文献

[1]
Hay-Wells syndrome in a child with mutation in the TP73L gene.

J Dtsch Dermatol Ges. 2007-10

[2]
An intermediate phenotype between Hay-Wells and Rapp-Hodgkin syndromes in a patient with a novel P63 mutation: confirmation of a variable phenotypic spectrum with a common aetiology.

Genet Couns. 2008

[3]
Rapp-Hodgkin and Hay-Wells ectodermal dysplasia syndromes represent a variable spectrum of the same genetic disorder.

Br J Dermatol. 2010-9

[4]
De novo missense mutation, S541Y, in the p63 gene underlying Rapp-Hodgkin ectodermal dysplasia syndrome.

Clin Exp Dermatol. 2005-5

[5]
Molecular evidence that AEC syndrome and Rapp-Hodgkin syndrome are variable expression of a single genetic disorder.

Clin Genet. 2004-7

[6]
Two novel TP63 mutations associated with the ankyloblepharon, ectodermal defects, and cleft lip and palate syndrome: a skin fragility phenotype.

Arch Dermatol. 2005-12

[7]
AEC syndrome: further evidence of a common genetic etiology with Rapp-Hodgkin syndrome.

Eur J Med Genet. 2006

[8]
Spectrum of p63 mutations in a selected patient cohort affected with ankyloblepharon-ectodermal defects-cleft lip/palate syndrome (AEC).

Am J Med Genet A. 2009-9

[9]
Variable expression in ankyloblepharon-ectodermal defects-cleft lip and palate syndrome.

Am J Med Genet. 1987-5

[10]
Ectodermal dysplasia showing clinical overlap between AEC, Rapp-Hodgkin and CHAND syndromes.

Clin Exp Dermatol. 2004-9

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