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XPC 基因频发突变 c.1643_1644delTG 的携带率与摩洛哥着色性干皮病的出生患病率。

Carrier frequency of the recurrent mutation c.1643_1644delTG in the XPC gene and birth prevalence of the xeroderma pigmentosum in Morocco.

机构信息

Center of Human Genomics, University Mohammed V Souissi Department of Medical Genetics, Morocco.

出版信息

J Dermatol. 2012 Apr;39(4):382-4. doi: 10.1111/j.1346-8138.2011.01453.x. Epub 2011 Dec 29.

Abstract

Xeroderma pigmentosum (XP) is an autosomal recessive inherited disease which is genetically heterogeneous. The prevalence of this genodermatosis is estimated to be 1/1 000 000 in the USA; it is more common in Japan and probably in other populations with high levels of consanguinity. The molecular diagnosis and identification of mutation in patients requires the knowledge of the causative gene by the determination of XP complementation groups. Soufir et al. have reported that XPC is the major disease-causing gene with a recurrent mutation in the Mediterranean region. The mutation c.1643_1644delTG (p.Val548AlafsX25) represents alone 74% of all the XP probands tested and 87% in XP type C in North African patients with founder effect. We used molecular epidemiological methods in the present study to calculate the frequency of heterozygote for this mutation in Moroccan newborns and estimate the prevalence of XP in the Moroccan population. DNA extracted from umbilical cord blood samples of 250 newborns were tested for the recurrent XPC mutation c.1643_1644delTG using real-time polymerase chain reaction. Heterozygotes profiles were confirmed by direct sequencing. Among 250 newborns tested, one subject was heterozygous for the mutation c.1643_1644delTG. The carrier frequency was estimated to be 1/250 which would imply that the prevalence of XP would be approximately 1/80 504 considering the effect of consanguinity. This is the first report of the prevalence of XP in an Arab country and it shows that the prevalence of xeroderma pigmentosum is higher than that found in Europe and the USA.

摘要

着色性干皮病(XP)是一种常染色体隐性遗传疾病,具有遗传异质性。据估计,这种遗传性皮肤病在美国的患病率为 1/1000000;在日本更为常见,在其他近亲结婚率较高的人群中可能也更为常见。要对患者进行分子诊断和突变识别,就需要通过确定 XP 互补组来了解致病基因。Soufir 等人报道称,XPC 是主要的致病基因,在地中海地区存在反复突变。c.1643_1644delTG(p.Val548AlafsX25)突变单独占所有受检 XP 先证者的 74%,在北非 XP 患者中占 XP 类型 C 的 87%,存在奠基者效应。本研究采用分子流行病学方法,计算摩洛哥新生儿杂合子的发生率,并估计摩洛哥人群中 XP 的患病率。采用实时聚合酶链反应,对 250 例新生儿脐带血样本中的 XPC 重复突变 c.1643_1644delTG 进行检测。通过直接测序对杂合子图谱进行确认。在 250 例受检新生儿中,有 1 例为 c.1643_1644delTG 突变的杂合子。携带者频率估计为 1/250,考虑到近亲结婚的影响,XP 的患病率约为 1/80504。这是阿拉伯国家首次报道 XP 的患病率,表明 XP 的患病率高于欧洲和美国。

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