| Literature DB >> 29569758 |
Ambreen Ijaz1,2, Sulman Basit3, Ajab Gul1, Lilas Batool1, Abrar Hussain1, Sibtain Afzal4, Khushnooda Ramzan5, Jamil Ahmad1, Abdul Wali1.
Abstract
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.Entities:
Keywords: Pakistani; XPC; autosomal recessive; founder mutation; xeroderma pigmentosum
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Year: 2018 PMID: 29569758 DOI: 10.1111/cga.12281
Source DB: PubMed Journal: Congenit Anom (Kyoto) ISSN: 0914-3505 Impact factor: 1.409