• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

囊性纤维化疾病中 CFTR 复合等位基因与表型变异性。

CFTR complex alleles and phenotypic variability in cystic fibrosis disease.

机构信息

Laboratory of Cellular and Molecular Genetics, Department of Genetics, Alexandria University, Aflaton Street- EL-Shatby, Alexandria 21545, Egypt.

Neurovascular Unit and Cognitive Disorders (NEUVACOD), University of Poitiers, Pôle Biologie Santé, Poitiers, France.

出版信息

Cell Mol Biol (Noisy-le-grand). 2024 Sep 8;70(8):244-260. doi: 10.14715/cmb/2024.70.8.33.

DOI:10.14715/cmb/2024.70.8.33
PMID:39262237
Abstract

Cystic fibrosis (CF) is inherited by CFTR (cystic fibrosis transmembrane conductance regulator) gene mutations. A variety of mutations have been identified in the CFTR gene that may be associated with cystic fibrosis, and these mutations demonstrate extensive molecular genetic heterogeneity in this disease. Little is known about the molecular mechanism by which mutations affect CFTR function, and only a minority of mutations have been characterized by functional studies. There has been an increase in the number of complex alleles. This may partly explain the difficulty in establishing genotype-phenotype correlations and complicate genetic counseling and diagnosis in some cases. Therefore, the identification of complex alleles has several important implications for recessive disorders. This will facilitate diagnosis; improve judgements concerning prognosis, and enable appropriate genetic counselling for affected families. This review describes the complex cystic fibrosis allele to better understand the contribution of this allele in the wide phenotypic variability of cystic fibrosis disease. It occurs in the complex allele that the second cis mutation can modulate the effects of the first mutation or vice versa. The phenotypic variability between CF or CFTR-RD (CFTR related disease) patients may be due to several factors, including different genetic and environmental backgrounds. It is important to determine the allele complex so that optimal treatment can be established.

摘要

囊性纤维化 (CF) 是由 CFTR(囊性纤维化跨膜电导调节因子)基因突变引起的。已经在 CFTR 基因中鉴定出多种可能与囊性纤维化相关的突变,这些突变在该疾病中表现出广泛的分子遗传异质性。关于突变如何影响 CFTR 功能的分子机制知之甚少,并且只有少数突变通过功能研究进行了表征。复杂等位基因的数量有所增加。这可能部分解释了建立基因型-表型相关性的困难,并在某些情况下使遗传咨询和诊断复杂化。因此,复杂等位基因的鉴定对隐性疾病具有重要意义。这将有助于诊断;改善预后判断,并为受影响的家庭提供适当的遗传咨询。本文综述了复杂的囊性纤维化等位基因,以更好地理解该等位基因在囊性纤维化疾病广泛表型变异性中的作用。它发生在第二个顺式突变可以调节第一个突变的作用或反之亦然的复杂等位基因中。CF 或 CFTR-RD(CFTR 相关疾病)患者之间的表型变异性可能是由于多种因素引起的,包括不同的遗传和环境背景。确定等位基因的复杂性很重要,这样才能建立最佳的治疗方案。

相似文献

1
CFTR complex alleles and phenotypic variability in cystic fibrosis disease.囊性纤维化疾病中 CFTR 复合等位基因与表型变异性。
Cell Mol Biol (Noisy-le-grand). 2024 Sep 8;70(8):244-260. doi: 10.14715/cmb/2024.70.8.33.
2
Genotype-phenotype correlation and functional studies in patients with cystic fibrosis bearing CFTR complex alleles.携带CFTR复合等位基因的囊性纤维化患者的基因型-表型相关性及功能研究。
J Med Genet. 2017 Apr;54(4):224-235. doi: 10.1136/jmedgenet-2016-103985. Epub 2016 Oct 13.
3
Identification and structural analysis for the first mutation in the cystic fibrosis transmembrane conductance regulator protein in Iran: case report and developmental insight using microsatellite markers.伊朗囊性纤维化跨膜电导调节蛋白中第一个突变的鉴定和结构分析:病例报告及微卫星标记的发育研究
Ther Adv Respir Dis. 2024 Jan-Dec;18:17534666241253990. doi: 10.1177/17534666241253990.
4
Things come in threes: A new complex allele and a novel deletion within the CFTR gene complicate an accurate diagnosis of cystic fibrosis.事不过三:CFTR 基因内的一个新的复杂等位基因和一个新的缺失使囊性纤维化的准确诊断变得复杂。
Mol Genet Genomic Med. 2022 Jun;10(6):e1926. doi: 10.1002/mgg3.1926. Epub 2022 Mar 29.
5
A new complex allele of the CFTR gene partially explains the variable phenotype of the L997F mutation.一种 CFTR 基因的新复合等位基因部分解释了 L997F 突变的可变表型。
Genet Med. 2010 Sep;12(9):548-55. doi: 10.1097/GIM.0b013e3181ead634.
6
A neutral variant involved in a complex CFTR allele contributes to a severe cystic fibrosis phenotype.一个复杂CFTR等位基因中的中性变异导致了严重的囊性纤维化表型。
Hum Genet. 2005 May;116(6):454-60. doi: 10.1007/s00439-004-1246-z. Epub 2005 Mar 3.
7
A Genotypic-Oriented View of CFTR Genetics Highlights Specific Mutational Patterns Underlying Clinical Macrocategories of Cystic Fibrosis.从基因型角度看CFTR基因遗传学,凸显了囊性纤维化临床大类背后的特定突变模式。
Mol Med. 2015 Apr 21;21(1):257-75. doi: 10.2119/molmed.2014.00229.
8
Complete and rapid scanning of the cystic fibrosis transmembrane conductance regulator (CFTR) gene by denaturing high-performance liquid chromatography (D-HPLC): major implications for genetic counselling.通过变性高效液相色谱法(D-HPLC)对囊性纤维化跨膜传导调节因子(CFTR)基因进行完整快速扫描:对遗传咨询的重要意义。
Hum Genet. 2001 Apr;108(4):290-8. doi: 10.1007/s004390100490.
9
Genotype and phenotype in cystic fibrosis.囊性纤维化中的基因型与表型。
Respiration. 2000;67(2):117-33. doi: 10.1159/000029497.
10
Cystic fibrosis transmembrane regulator haplotypes in households of patients with cystic fibrosis.囊性纤维化患者家庭中的囊性纤维化跨膜传导调节因子单倍型
Gene. 2018 Jan 30;641:137-143. doi: 10.1016/j.gene.2017.10.052. Epub 2017 Oct 18.

引用本文的文献

1
-Regulation of the Gene in Pancreatic Cells.-胰腺细胞中基因的调控。
Int J Mol Sci. 2025 Apr 17;26(8):3788. doi: 10.3390/ijms26083788.