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[家族性主动脉瓣上狭窄。一个家族的调查及文献综述]

[Familial supravalvular aortic stenosis. Investigation in a family and review of the literature].

作者信息

Burnel P, Marçon F, Lucron H, Bosser G, Gilgenkrantz S, Jonveaux P, Chéry M, Worms A M

机构信息

Service de cardiologie infantile, hôpital d'Enfants, Vandoeuvre-lés-Nancy.

出版信息

Arch Mal Coeur Vaiss. 1997 May;90(5):719-24.

PMID:9295957
Abstract

Familial supravalvular aortic stenosis is a rare autosomal dominant condition. It may be distinguished from the Williams-Beuren syndrome by the absence of the characteristic dysmorphic appearances and of mental retardation. The case of a 9-year-old girl with a severe surgical stenosis led to the diagnosis of the same malformation in the mother and two brothers. This family adds to the 121 cases reported in the literature describing the main features of SVAS. Molecular biological advances have shown that familial SVAS and the Williams syndrome are due to mutation of the elastin gene located at 7q11-23. In the Williams syndrome the allele of this gene is completely absent and there is also probably deletion of contiguous genes, which explains involvement of cognitive function. In SVAS, the genetic lesion, mutation or microdeletion is more limited, explaining the usually isolated aortic malformation. Other studies are necessary to confirm these results.

摘要

家族性主动脉瓣上狭窄是一种罕见的常染色体显性疾病。它可通过缺乏特征性的畸形外观和智力发育迟缓与威廉姆斯-贝伦综合征相鉴别。一名患有严重手术性狭窄的9岁女孩的病例,导致其母亲和两个兄弟被诊断出患有相同的畸形。这个家族补充了文献中报道的121例描述主动脉瓣上狭窄主要特征的病例。分子生物学进展表明,家族性主动脉瓣上狭窄和威廉姆斯综合征是由于位于7q11-23的弹性蛋白基因突变所致。在威廉姆斯综合征中,该基因的等位基因完全缺失,并且可能还存在相邻基因的缺失,这解释了认知功能的受累情况。在主动脉瓣上狭窄中,基因损伤、突变或微缺失更为局限,这解释了通常仅存在主动脉畸形的情况。需要进行其他研究来证实这些结果。

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