Ijaz Ambreen, Basit Sulman, Gul Ajab, Batool Lilas, Hussain Abrar, Afzal Sibtain, Ramzan Khushnooda, Ahmad Jamil, Wali Abdul
Department of Biotechnology, Faculty of Life Sciences & Informatics, BUITEMS, Quetta, Pakistan.
Department of Zoology, Sardar Bahadur Khan Women's University, Quetta, Pakistan.
Congenit Anom (Kyoto). 2019 Jan;59(1):18-21. doi: 10.1111/cga.12281. Epub 2018 Apr 15.
Xeroderma pigmentosum (XP) is a rare autosomal recessive skin disorder characterized by hyperpigmentation, premature skin aging, ocular and cutaneous photosensitivity, and increased risk of skin carcinoma. We investigated seven consanguineous XP families with nine patients from Pakistan. All the Patients exhibited typical clinical symptoms of XP since first year of life. Whole genome SNP genotyping identified a 14 Mb autozygous region segregating with the disease phenotype on chromosome 3p25.1. DNA sequencing of XPC gene revealed a founder homozygous splice site mutation (c.2251-1G>C) in patients from six families (A-F) and a homozygous nonsense mutation (c.1399C>T; p.Gln467*) in patients of family G. This is the first report of XPC mutations, underlying XP phenotype, in Pakistani population.
着色性干皮病(XP)是一种罕见的常染色体隐性皮肤病,其特征为色素沉着过度、皮肤过早老化、眼部和皮肤对光敏感以及皮肤癌风险增加。我们对来自巴基斯坦的7个近亲XP家族中的9名患者进行了研究。所有患者自出生第一年起就表现出典型的XP临床症状。全基因组单核苷酸多态性(SNP)基因分型确定了一个14兆碱基的纯合区域,该区域与3号染色体p25.1上的疾病表型共分离。XPC基因的DNA测序显示,6个家族(A - F)的患者存在一个始祖纯合剪接位点突变(c.2251-1G>C),而家族G的患者存在一个纯合无义突变(c.1399C>T;p.Gln467*)。这是巴基斯坦人群中导致XP表型的XPC突变的首次报道。