Molecular Investigation of Genetic Orphan Diseases' Research Unit, Pasteur Institute of Tunis, Tunis, Tunisia.
J Hum Genet. 2009 Jul;54(7):426-9. doi: 10.1038/jhg.2009.50. Epub 2009 May 29.
Xeroderma pigmentosum (XP, OMIM 278700-278780) is a group of autosomal recessive diseases characterized by hypersensitivity to UV rays. There are seven complementation groups of XP (XPA to XPG) and XPV. Among them, the XP group C (XP-C) is the most prevalent type in Western Europe and in the United States. We report here on the clinical and genetic investigation of XP-C patients in 14 Tunisian families. As the XPC V548A fs X572 mutation has been identified in Algerian and Moroccan populations, Tunisian patients were first screened for this mutation by a direct sequencing of exon 9 of the XPC gene. All patients with a severe clinical form had this mutation, thus showing the homogeneity of the mutational spectrum of XPC in Tunisia. A potential founder effect was searched and confirmed by haplotype analysis. Taking into account the similarity of the genetic background, we propose a direct screening of this mutation as a rapid and cost-effective tool for the diagnosis of XP-C in North Africa.
着色性干皮病(XP,OMIM 278700-278780)是一组常染色体隐性疾病,其特征为对紫外线过敏。XP 有七个互补群(XPA 至 XPG)和 XPV。其中,XP 组 C(XP-C)在西欧和美国最为常见。我们在此报告了对 14 个突尼斯家庭的 XP-C 患者的临床和遗传调查。由于 XPC V548A fs X572 突变已在阿尔及利亚和摩洛哥人群中被鉴定出来,因此首先通过直接测序 XPC 基因的第 9 外显子对突尼斯患者进行了该突变的筛查。所有具有严重临床形式的患者均具有该突变,因此显示了突尼斯 XPC 突变谱的同质性。通过单体型分析搜索并确认了潜在的创始人效应。考虑到遗传背景的相似性,我们建议直接筛查该突变作为北非 XP-C 快速且具有成本效益的诊断工具。