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