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P基因122.5千碱基的缺失是纳瓦霍人群中2型眼皮肤白化病高患病率的根本原因。

A 122.5-kilobase deletion of the P gene underlies the high prevalence of oculocutaneous albinism type 2 in the Navajo population.

作者信息

Yi Zanhua, Garrison Nanibaa', Cohen-Barak Orit, Karafet Tatiana M, King Richard A, Erickson Robert P, Hammer Michael F, Brilliant Murray H

机构信息

Department of Pediatrics, College of Medicine, University of Arizona, Tucson, AZ 85724, USA.

出版信息

Am J Hum Genet. 2003 Jan;72(1):62-72. doi: 10.1086/345380. Epub 2002 Dec 5.

Abstract

Oculocutaneous albinism (OCA) is a genetically heterogeneous disorder. There are four known types of OCA: OCA1-OCA4. The clinical manifestations of all types of OCA include skin and hair hypopigmentation and visual impairment. Although there are a few documented observations of high frequency of albinism among Native Americans, including the Hopi, Zuni, Kuna, Jemez, Laguna, San Juan, and Navajo, no causative molecular defect has been previously reported. In the present study, we show that albinism in one Native American population, the Navajo, is caused by a LINE-mediated 122.5-kilobase deletion of the P gene, thus demonstrating that albinism in this population is OCA2. This deletion appears to be Navajo specific, because this allele was not detected in 34 other individuals with albinism who listed other Native American origins, nor has it been reported in any other ethnic group. The molecular characterization of this deletion allele allowed us to design a three-primer polymerase chain reaction system to estimate the carrier frequency in the Navajo population by screening 134 unrelated normally pigmented Navajos. The carrier frequency was found to be approximately 4.5%. The estimated prevalence of OCA2 in Navajos is between approximately 1 per 1,500 and 1 per 2,000. We further estimate that this mutation originated 400-1,000 years ago from a single founder.

摘要

眼皮肤白化病(OCA)是一种基因异质性疾病。已知有四种类型的OCA:OCA1 - OCA4。所有类型的OCA的临床表现包括皮肤和毛发色素减退以及视力损害。尽管有一些文献记载了美洲原住民(包括霍皮人、祖尼人、库纳人、杰梅兹人、拉古纳人、圣胡安人和纳瓦霍人)中白化病的高发病率,但此前尚未报道其致病分子缺陷。在本研究中,我们表明美洲原住民纳瓦霍人群中的白化病是由LINE介导的P基因122.5千碱基缺失引起的,从而证明该人群中的白化病是OCA2。这种缺失似乎是纳瓦霍人特有的,因为在其他列出美洲原住民起源的34名白化病个体中未检测到该等位基因,在任何其他种族群体中也未报道过。对这种缺失等位基因的分子特征分析使我们能够设计一种三引物聚合酶链反应系统,通过筛查134名无亲缘关系的正常色素沉着的纳瓦霍人来估计纳瓦霍人群中的携带者频率。发现携带者频率约为4.5%。纳瓦霍人中OCA2的估计患病率约为每1500人中有1例至每2000人中有1例。我们进一步估计,这种突变起源于400 - 1000年前的一个单一始祖。

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