Mota Laís Ribeiro, de Melo Filho Valmir Machado, de Castro Lorena Lemos, Garcia Daniel Fantozzi, Terse-Ramos Regina, Toralles Maria Betânia Pereira, de Lima Renata Lúcia Leite Ferreira, Souza Edna Lúcia
Post-Graduation Program in Interactive Processes of Systems and Organs, Institute of Health Sciences, Federal University of Bahia, Salvador, Brazil.
School of Medicine of Bahia, Federal University of Bahia, Salvador, Brazil.
Mol Biol Rep. 2018 Dec;45(6):2045-2051. doi: 10.1007/s11033-018-4361-y. Epub 2018 Sep 19.
Knowledge of the genetic profile of Cystic Fibrosis (CF) contributes to a better understanding of the genotype/phenotype relationship, particularly in mixed populations such as in Brazil. To describe clinical data of CF patients with rare or not yet observed CFTR gene mutations in Brazil. It was a case series of CF patients followed-up at a referral center. Clinical and laboratory data were obtained through medical records. Molecular analysis of the mutations was performed by conventional methods and/or by next-generation sequencing. Ten patients were studied, seven had five pathogenic mutations without previous description in Brazil (Q1100P, Y109C, A107P, E1409K and K162E), one of which has not yet been reported in patients with CF (A107P). Among the seven patients, three (two siblings) had the second mutant allele of rare occurrence among Brazilians patients (G1069R and 2307insA). Three other patients also had at least one rare variant (V201M, S466X and G1069R). The age of the CF diagnosis ranged from 1 to 190 months in the ten cases and the main clinical manifestations were respiratory symptoms and difficulty in gaining weight. All but one patient presented clinical and/or laboratory data compatible with pancreatic insufficiency. The identification of rare or not yet described CFTR mutations in patients with CF in Brazil highlights the high genetic heterogeneity in this population. Knowledge of the genotypic profile of Brazilian CF patients can contribute to the development of specific mutation panels for the genetic investigation targeting each region of the country, as well as helping to understand the complex genotype/phenotype relationship, especially in mixed populations.
了解囊性纤维化(CF)的基因概况有助于更好地理解基因型/表型关系,特别是在巴西这样的混合人群中。描述巴西患有罕见或尚未观察到的CFTR基因突变的CF患者的临床数据。这是在一家转诊中心对CF患者进行随访的病例系列研究。通过病历获取临床和实验室数据。通过传统方法和/或下一代测序对突变进行分子分析。研究了10名患者,其中7名有5种致病性突变,此前在巴西没有描述(Q1100P、Y109C、A107P、E1409K和K162E),其中一种在CF患者中尚未报道(A107P)。在这7名患者中,3名(2名是兄弟姐妹)具有在巴西患者中罕见的第二个突变等位基因(G1069R和2307insA)。另外3名患者也至少有一种罕见变异(V201M、S466X和G1069R)。这10例患者的CF诊断年龄为1至190个月,主要临床表现为呼吸道症状和体重增加困难。除1名患者外,所有患者均呈现与胰腺功能不全相符的临床和/或实验室数据。在巴西CF患者中鉴定出罕见或尚未描述的CFTR突变,突出了该人群中高度的基因异质性。了解巴西CF患者的基因型概况有助于开发针对该国每个地区的基因检测特定突变组合,也有助于理解复杂的基因型/表型关系,特别是在混合人群中。