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肺动脉高压突变谱与苏北人群苯丙酮尿症表现的相关性。

PAH mutation spectrum and correlation with PKU manifestation in north Jiangsu province population.

机构信息

Department of Medical Laboratory, Lianyungang Maternal and Child Health Hospital, Lianyungang, Jiangsu, China.

Department of Biomedical Science, Mercer University School of Medicine, Macon, GA, USA.

出版信息

Kaohsiung J Med Sci. 2018 Feb;34(2):89-94. doi: 10.1016/j.kjms.2017.09.006. Epub 2017 Oct 5.

Abstract

Phenylketonuria (PKU) is a common autosomal recessive disorder of phenylalanine metabolism and mainly results a deficiency of phenylalanine hydroxylase gene (PAH). The incidence of various PAH mutations have race and ethnicity differences. We report a spectrum of PAH mutations complied from 35 PKU children who are all Chinese Han population from north Jiangsu in this study. All 13 exons and their flanking intron sequences of PAH were determined by Ion Torrent PGM™ sequencing. The relationship of genotype and phenotype was analyzed based on the sum of the arbitrary value (AV) values of the two alleles. We identified 61 mutations, with a frequency of 87.14%, among 70 alleles of 35 patients. The most prevalent mutations were R243Q (26.23%), R241C (9.84%) and V399V (8.20%). Furthermore, the consistency between prediction of the biochemical phenotype and the observed phenotype was 81.25%, with the highest consistency observed in classic PKU (87.50%). A significant correlation was found between pretreatment levels of phenylalanine and AV sum (r = -0.87, P < 0.05). Finally, our study constructs PAH mutation spectrum by next generation sequencing (NGS), and reveals that the PAH genotypes and biochemical phenotypes were significantly correlated. These offers facilitate the provision of appropriate genetic counseling for PKU patients.

摘要

苯丙酮尿症(PKU)是一种常见的常染色体隐性遗传疾病,主要是由于苯丙氨酸羟化酶基因(PAH)缺乏所致。各种 PAH 突变的发生率存在种族和民族差异。我们报告了来自苏北的 35 名 PKU 儿童的 PAH 突变谱,他们均为汉族。通过 Ion Torrent PGM™测序确定了 PAH 的 13 个外显子及其侧翼内含子序列。基于两个等位基因的任意值(AV)值之和,分析了基因型和表型之间的关系。我们在 35 名患者的 70 个等位基因中鉴定出 61 种突变,频率为 87.14%。最常见的突变是 R243Q(26.23%),R241C(9.84%)和 V399V(8.20%)。此外,生化表型的预测与观察表型之间的一致性为 81.25%,经典 PKU 的一致性最高(87.50%)。治疗前苯丙氨酸水平与 AV 总和之间存在显著相关性(r = -0.87,P <0.05)。最后,我们通过下一代测序(NGS)构建了 PAH 突变谱,并揭示了 PAH 基因型和生化表型之间存在显著相关性。这些为 PKU 患者提供了适当的遗传咨询提供了便利。

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