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一个具有沃森综合征和努南综合征特征的家族中,1型神经纤维瘤病(NF1)基因外显子内的串联重复。

Tandem duplication within a neurofibromatosis type 1 (NF1) gene exon in a family with features of Watson syndrome and Noonan syndrome.

作者信息

Tassabehji M, Strachan T, Sharland M, Colley A, Donnai D, Harris R, Thakker N

机构信息

University Department of Medical Genetics, St. Mary's Hospital, Manchester, United Kingdom.

出版信息

Am J Hum Genet. 1993 Jul;53(1):90-5.

Abstract

Type 1 neurofibromatosis (NF1), Watson syndrome (WS), and Noonan syndrome (NS) show some overlap in clinical manifestations. In addition, WS has been shown to be linked to markers flanking the NF1 locus and a deletion at the NF1 locus demonstrated in a WS patient. This suggests either that WS and NF1 are allelic or that phenotypes arise from mutations in very closely linked genes. Here we provide evidence for the former by demonstrating a mutation in the NF1 gene in a family with features of both WS and NS. The mutation is an almost perfect in-frame tandem duplication of 42 bases in exon 28 of the NF1 gene. Unlike the mutations previously described in classical NF1, which show a preponderance of null alleles, the mutation in this family would be expected to result in a mutant neurofibromin product.

摘要

1型神经纤维瘤病(NF1)、沃森综合征(WS)和努南综合征(NS)在临床表现上存在一些重叠。此外,研究表明WS与NF1基因座侧翼的标记有关,并且在一名WS患者中发现了NF1基因座的缺失。这表明WS和NF1要么是等位基因,要么是表型由紧密连锁基因中的突变产生。在这里,我们通过在一个具有WS和NS特征的家族中证明NF1基因存在突变,为前者提供了证据。该突变是NF1基因第28外显子中42个碱基几乎完美的框内串联重复。与先前在经典NF1中描述的突变不同,经典NF1中的突变主要为无效等位基因,而这个家族中的突变预计会产生突变型神经纤维瘤蛋白产物。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/08df/1682238/8f8922eabcfa/ajhg00052-0097-a.jpg

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