Suppr超能文献

泰国皮杰氏综合征患者 STK11 基因的新突变。

Novel mutations in the STK11 gene in Thai patients with Peutz-Jeghers syndrome.

机构信息

Interdepartment of Biomedical Sciences, Faculty of Graduate School, Chulalongkorn University, and Department of Peidatrics, King Chulalongkorn Memorial Hospital, Bangkok, Thailand.

出版信息

World J Gastroenterol. 2009 Nov 14;15(42):5364-7. doi: 10.3748/wjg.15.5364.

Abstract

Peutz-Jeghers syndrome (PJS), a rare autosomal dominant inherited disorder, is characterized by hamartomatous gastrointestinal polyps and mucocutaneous pigmentation. Patients with this syndrome have a predisposition to a variety of cancers in multiple organs. Mutations in the serine/threonine kinase 11 (STK11) gene have been identified as a major cause of PJS. Here we present the clinical and molecular findings of two unrelated Thai individuals with PJS. Mutation analysis by Polymerase Chain Reaction-sequencing of the entire coding region of STK11 revealed two potentially pathogenic mutations. One harbored a single nucleotide deletion (c.182delG) in exon 1 resulting in a frameshift leading to premature termination at codon 63 (p.Gly61AlafsX63). The other carried an in-frame 9-base-pair (bp) deletion in exon 7, c.907_915del9 (p.Ile303_Gln305del). Both deletions were de novo and have never been previously described. This study has expanded the genotypic spectrum of the STK11 gene.

摘要

皮杰特-杰格斯综合征(PJS)是一种罕见的常染色体显性遗传疾病,其特征为错构瘤性胃肠道息肉和黏膜皮肤色素沉着。该综合征患者易发生多种器官的癌症。丝氨酸/苏氨酸激酶 11(STK11)基因突变已被确定为 PJS 的主要病因。本研究报告了两例无亲缘关系的泰国 PJS 患者的临床和分子学发现。通过聚合酶链反应-测序对 STK11 全长编码区进行突变分析,发现了两种潜在的致病性突变。一种携带 1 个核苷酸缺失(c.182delG),导致外显子 1 移码,从而在密码子 63 提前终止(p.Gly61AlafsX63)。另一种携带 7 个碱基对(bp)的框内缺失,c.907_915del9(p.Ile303_Gln305del)。这两种缺失均为新生突变,此前从未被描述过。本研究扩展了 STK11 基因突变谱。

相似文献

本文引用的文献

1
The molecular mechanisms that underlie the tumor suppressor function of LKB1.LKB1肿瘤抑制功能背后的分子机制。
Acta Biochim Biophys Sin (Shanghai). 2009 Feb;41(2):97-107. doi: 10.1093/abbs/gmn011.
3
Frequency and spectrum of cancers in the Peutz-Jeghers syndrome.佩-吉综合征中癌症的发生率及谱型
Clin Cancer Res. 2006 May 15;12(10):3209-15. doi: 10.1158/1078-0432.CCR-06-0083.
5
Mutations in the human LKB1/STK11 gene.人类LKB1/STK11基因的突变
Hum Mutat. 2005 Oct;26(4):291-7. doi: 10.1002/humu.20222.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验