Garcia Bartels Natalie, Neumann Luitgard M, Mleczko Anna, Rubach Katharina, Peters Hartmut, Rossi Rainer, Sterry Wolfram, Blume-Peytavi Ulrike
Department of Dermatology and Allergy, Charité- Universitätsmedizin Berlin, Germany.
J Dtsch Dermatol Ges. 2007 Oct;5(10):919-23. doi: 10.1111/j.1610-0387.2007.06379.x.
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.
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。
J Dtsch Dermatol Ges. 2007-10
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