Alibakhshi Reza, Moradi Keyvan, Mohebbi Zahra, Ghadiri Keyghobad
Department of Biochemistry, School of Medicine, Kermanshah University of Medical Sciences, Kermanshah, Iran,
Metab Brain Dis. 2014 Mar;29(1):131-8. doi: 10.1007/s11011-013-9432-0. Epub 2013 Sep 19.
Phenylketonuria (PKU) is an autosomal recessive disorder characterized by a mutation in the phenylalanine hydroxylase (PAH) gene. Untreated PKU can lead to mental retardation, seizures, and other serious medical problems. This study was designed to investigate the status of molecular defects in the PAH gene and their association with polymorphisms in Kurdish patients with PKU in the Kermanshah province, western Iran. The study was conducted on 27 unrelated patients with PKU over a 2-year period (from 2010 to 2012). All 13 exons plus exon-intron boundaries of the PAH gene were analyzed and we identified 15 different mutations, including two novel mutations, in 51 of the 54 mutant alleles (diagnostic efficiency of 94.4 %). IVS4 + 1G > C (c.441 + 1G > C) and IVS7 - 5 T > C (c.843 - 5 T > C) are novel mutations that have not been reported in the academic literature or the PAH locus database ( http://www.pahdb.mcgill.ca ); therefore, they may be specific to the Kurdish population. IVS2 + 5G > C and IVS9 + 5G > A were the two most prevalent mutations in our sample, with frequencies of 26 % and 17 %, respectively. The second most common mutations were p.R261X, IVS10 - 11G > A, p.K363 > Nfs and IVS7 - 5 T > C, with each showing a relative frequency of 7.4 %. All other detected mutations, including p.F55 > Lfs, p.R176X, p.R243Q, p.V230I, p.R243X, p.R261Q, IVS8 - 7A > G and p.E390G had frequencies of less than 4 %. The present study showed that there is a distinct difference in the characteristics of PAH mutations between the Kermanshah province and other parts of Iran, suggesting that Kermanshah may have a unique population distribution of PAH gene mutations. Iran lies on the route of major ancient movements of the Caucasian people toward the Mediterranean basin, and Kermanshah has previously been called the gateway to Asia. Most of the mutations identified in this study are common in the Mediterranean region. Therefore, our findings are consistent with the historical and geographical links between the Iranian population and the populations of Mediterranean region.
苯丙酮尿症(PKU)是一种常染色体隐性疾病,其特征为苯丙氨酸羟化酶(PAH)基因突变。未经治疗的PKU可导致智力发育迟缓、癫痫发作及其他严重的医学问题。本研究旨在调查伊朗西部克尔曼沙阿省PKU库尔德患者中PAH基因的分子缺陷状况及其与多态性的关联。该研究在2年期间(2010年至2012年)对27例无亲缘关系的PKU患者进行。对PAH基因的所有13个外显子及外显子 - 内含子边界进行了分析,我们在54个突变等位基因中的51个中鉴定出15种不同突变,包括两种新突变(诊断效率为94.4%)。IVS4 + 1G > C(c.441 + 1G > C)和IVS7 - 5 T > C(c.843 - 5 T > C)是新突变,学术文献或PAH基因座数据库(http://www.pahdb.mcgill.ca)中均未报道;因此,它们可能是库尔德人群特有的。IVS2 + 5G > C和IVS9 + 5G > A是我们样本中最常见的两种突变,频率分别为26%和17%。第二常见的突变是p.R261X、IVS10 - 11G > A、p.K363 > Nfs和IVS7 - 5 T > C,相对频率均为7.4%。所有其他检测到的突变,包括p.F55 > Lfs、p.R176X、p.R243Q、p.V230I、p.R243X、p.R261Q、IVS8 - 7A > G和p.E390G,频率均低于4%。本研究表明,克尔曼沙阿省与伊朗其他地区的PAH突变特征存在明显差异,这表明克尔曼沙阿可能具有独特的PAH基因突变人群分布。伊朗位于高加索人向地中海盆地主要古代迁徙路线上,克尔曼沙阿此前被称为亚洲之门。本研究中鉴定出的大多数突变在地中海地区很常见。因此,我们的研究结果与伊朗人群和地中海地区人群之间的历史和地理联系一致。