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欧洲苯丙酮尿症的多种起源。

Multiple origins for phenylketonuria in Europe.

作者信息

Eisensmith R C, Okano Y, Dasovich M, Wang T, Güttler F, Lou H, Guldberg P, Lichter-Konecki U, Konecki D S, Svensson E

机构信息

Howard Hughes Medical Institute, Department of Cell Biology, Baylor College of Medicine, Houston, Texas 77030.

出版信息

Am J Hum Genet. 1992 Dec;51(6):1355-65.

Abstract

Phenylketonuria (PKU), a disorder of amino acid metabolism prevalent among Caucasians and other ethnic groups, is caused primarily by a deficiency of the hepatic enzyme phenylalanine hydroxylase (PAH). PKU is a highly heterogeneous disorder, with more than 60 molecular lesions identified in the PAH gene. The haplotype associations, relative frequencies, and distributions of five prevalent PAH mutations (R158Q, R261Q, IVS10nt546, R408W, and IVS12n1) were established in a comprehensive European sample population and subsequently were examined to determine the potential roles of several genetic mechanisms in explaining the present distribution of the major PKU alleles. Each of these five mutations was strongly associated with only one of the more than 70 chromosomal haplotypes defined by eight RFLPs in or near the PAH gene. These findings suggest that each of these mutations arose through a single founding event that occurred within time periods ranging from several hundred to several thousand years ago. From the significant differences observed in the relative frequencies and distributions of these five alleles throughout Europe, four of these putative founding events could be localized to specific ethnic subgroups. Together, these data suggest that there were multiple, geographically and ethnically distinct origins for PKU within the European population.

摘要

苯丙酮尿症(PKU)是一种在高加索人和其他种族群体中普遍存在的氨基酸代谢紊乱疾病,主要由肝脏酶苯丙氨酸羟化酶(PAH)缺乏引起。PKU是一种高度异质性疾病,在PAH基因中已鉴定出60多种分子病变。在一个全面的欧洲样本群体中确定了五种常见PAH突变(R158Q、R261Q、IVS10nt546、R408W和IVS12n1)的单倍型关联、相对频率和分布,随后对其进行了研究,以确定几种遗传机制在解释主要PKU等位基因当前分布中的潜在作用。这五种突变中的每一种都仅与PAH基因内或附近由八个限制性片段长度多态性(RFLP)定义的70多种染色体单倍型中的一种强烈相关。这些发现表明,这些突变中的每一种都是通过一次单一的奠基事件产生的,这些事件发生在几百年到几千年前的时间段内。从在整个欧洲观察到的这五个等位基因的相对频率和分布的显著差异来看,其中四个假定的奠基事件可以定位到特定的种族亚群。总之,这些数据表明欧洲人群中PKU有多个地理和种族上不同的起源。

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