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马凡综合征家族中的产前诊断及原纤维蛋白的供体剪接位点突变

Prenatal diagnosis and a donor splice site mutation in fibrillin in a family with Marfan syndrome.

作者信息

Godfrey M, Vandemark N, Wang M, Velinov M, Wargowski D, Tsipouras P, Han J, Becker J, Robertson W, Droste S

机构信息

Department of Pediatrics, University of Nebraska Medical Center, Omaha 68198.

出版信息

Am J Hum Genet. 1993 Aug;53(2):472-80.

Abstract

The Marfan syndrome, an autosomal dominant connective tissue disorder, is manifested by abnormalities in the cardiovascular, skeletal, and ocular systems. Recently, fibrillin, an elastin-associated microfibrillar glycoprotein, has been linked to the Marfan syndrome, and fibrillin mutations in affected individuals have been documented. In this study, genetic linkage analysis with fibrillin specific markers was used to establish the prenatal diagnosis in an 11-wk-gestation fetus in a four-generation Marfan kindred. At birth, skeletal changes suggestive of the Marfan syndrome were observed. Reverse transcription-PCR amplification of the fibrillin gene mRNA detected a deletion of 123 bp in one allele in affected relatives. This deletion corresponds to an exon encoding an epidermal growth factor-like motif. Examination of genomic DNA showed a G-->C transversion at the +1 consensus donor splice site.

摘要

马方综合征是一种常染色体显性遗传性结缔组织疾病,表现为心血管、骨骼和眼部系统异常。最近,原纤维蛋白,一种与弹性蛋白相关的微纤维糖蛋白,已与马方综合征联系起来,并且已记录了受影响个体中的原纤维蛋白突变。在本研究中,利用原纤维蛋白特异性标记进行基因连锁分析,对一个四代马方家族中妊娠11周的胎儿进行产前诊断。出生时,观察到提示马方综合征的骨骼变化。对原纤维蛋白基因mRNA进行逆转录-聚合酶链反应扩增,在受影响亲属的一个等位基因中检测到123bp的缺失。该缺失对应于一个编码表皮生长因子样基序的外显子。对基因组DNA的检测显示,在+1共有供体剪接位点处发生了G→C颠换。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/11a3/1682369/113e544e1151/ajhg00053-0181-a.jpg

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