囊性纤维化疾病中的 CFTR 复合物等位基因和表型变异。

IF 1.5 4区 生物学 Q4 BIOCHEMISTRY & MOLECULAR BIOLOGY Cellular and molecular biology Pub Date : 2024-09-08 DOI:10.14715/cmb/2024.70.8.33
Ayman El-Seedy, Véronique Ladeveze
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引用次数: 0

摘要

囊性纤维化(CF)是通过 CFTR(囊性纤维化跨膜传导调节器)基因突变遗传的。目前已发现多种可能与囊性纤维化有关的 CFTR 基因突变,这些突变显示了该疾病广泛的分子遗传异质性。人们对基因突变影响 CFTR 功能的分子机制知之甚少,只有少数基因突变通过功能研究得到了定性。复杂等位基因的数量有所增加。这可能是难以确定基因型与表型相关性的部分原因,在某些情况下也会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遗传疾病具有重要意义。这将有助于诊断,改善对预后的判断,并为受影响的家庭提供适当的遗传咨询。本综述介绍了囊性纤维化复杂等位基因,以更好地了解该等位基因在囊性纤维化疾病的广泛表型变异中的作用。在复杂等位基因中,第二个顺式突变可以调节第一个突变的影响,反之亦然。CF 或 CFTR-RD(CFTR 相关疾病)患者之间的表型变异可能由多种因素造成,包括不同的遗传和环境背景。确定等位基因复合物非常重要,这样才能确定最佳治疗方法。
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CFTR complex alleles and phenotypic variability in cystic fibrosis disease.

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.

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来源期刊
Cellular and molecular biology
Cellular and molecular biology 生物-生化与分子生物学
CiteScore
1.60
自引率
12.50%
发文量
331
期刊介绍: Cellular and Molecular Biology publishes original articles, reviews, short communications, methods, meta-analysis notes, letters to editor and comments in the interdisciplinary science of Cellular and Molecular Biology linking and integrating molecular biology, biophysics, biochemistry, enzymology, physiology and biotechnology in a dynamic cell and tissue biology environment, applied to human, animals, plants tissues as well to microbial and viral cells. The journal Cellular and Molecular Biology is therefore open to intense interdisciplinary exchanges in medical, dental, veterinary, pharmacological, botanical and biological researches for the demonstration of these multiple links.
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