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[RAS/丝裂原活化蛋白激酶信号转导通路及其在努南综合征发病机制中的作用]

[RAS/MAPK signal transduction pathway and its role in the pathogenesis of Noonan syndrome].

作者信息

Gos Monika, Leszkiewicz Monika, Abramowicz Anna

机构信息

Zakład Genetyki Medycznej, Instytut Matki i Dziecka, Warszawa.

出版信息

Postepy Biochem. 2012;58(3):255-64.

Abstract

Noonan syndrome (NS) is one of the most frequent dysmorphic syndromes in children with a frequency of 1/1000-1/2500 of newborns. Noonan syndrome is a multi-organ disease with a broad spectrum of clinical symptoms. The most characteristic features of NS are: craniofacial dysmorphy, short stature, cardiovascular defects, bone and skeletal defects and delayed puberty (cryptorchidism in males). Noonan syndrome has a genetic background and is inherited in autosomal dominant manner. The recent studies have shown that it is due to the presence of mutation in one of the genes encoding proteins of RAS/MAPK signalling pathway responsible for cell proliferation and differentiation. Till now, NS causing mutations were identified in PTPN11, SOS1, RAF1, KRAS, BRAF, SHOC2 and NRAS genes, and this may partially explain the broad phenotypic spectrum observed in patients. Noonan syndrome is one of the RAS-opathies, therefore the molecular analysis of RAS/ MAPK genes might be a very useful tool in clinical differentiation of the disease.

摘要

努南综合征(NS)是儿童中最常见的畸形综合征之一,新生儿发病率为1/1000 - 1/2500。努南综合征是一种多器官疾病,临床症状广泛。NS最典型的特征是:颅面部畸形、身材矮小、心血管缺陷、骨骼和骨骼缺陷以及青春期延迟(男性隐睾)。努南综合征具有遗传背景,以常染色体显性方式遗传。最近的研究表明,这是由于负责细胞增殖和分化的RAS/MAPK信号通路蛋白编码基因之一发生突变所致。到目前为止,在PTPN11、SOS1、RAF1、KRAS、BRAF、SHOC2和NRAS基因中鉴定出了导致NS的突变,这可能部分解释了患者中观察到的广泛表型谱。努南综合征是RAS病之一,因此RAS/MAPK基因的分子分析可能是该疾病临床鉴别诊断中非常有用的工具。

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