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着色性干皮病:巴西着色性干皮病患者群体中种系XPA突变的低发生率

Xeroderma pigmentosum: low prevalence of germline XPA mutations in a Brazilian XP population.

作者信息

Santiago Karina Miranda, França de Nóbrega Amanda, Rocha Rafael Malagoli, Rogatto Silvia Regina, Achatz Maria Isabel

机构信息

International Research Center, A.C. Camargo Cancer Center, São Paulo, SP 01508-010, Brazil.

Department of Oncogenetics, A.C. Camargo Cancer Center, São Paulo, SP 01509-900, Brazil.

出版信息

Int J Mol Sci. 2015 Apr 22;16(4):8988-96. doi: 10.3390/ijms16048988.

Abstract

Xeroderma pigmentosum (XP) is a rare autosomal recessive disorder characterized by DNA repair defects that cause photophobia, sunlight-induced cancers, and neurodegeneration. Prevalence of germline mutations in the nucleotide excision repair gene XPA vary significantly in different populations. No Brazilian patients have been reported to carry a germline mutation in this gene. In this study, the germline mutational status of XPA was determined in Brazilian patients exhibiting major clinical features of XP syndrome. The study was conducted on 27 unrelated patients from select Brazilian families. A biallelic inactivating transition mutation c.619C>T (p.Arg207Ter) was identified in only one patient with a history of neurological impairment and mild skin abnormalities. These findings suggest that XP syndrome is rarely associated with inherited disease-causing XPA mutations in the Brazilian population. Additionally, this report demonstrates the effectiveness of genotype-phenotype correlation as a valuable tool to guide direct genetic screening.

摘要

着色性干皮病(XP)是一种罕见的常染色体隐性疾病,其特征是DNA修复缺陷,可导致畏光、阳光诱发的癌症和神经退行性变。核苷酸切除修复基因XPA中的种系突变在不同人群中的患病率差异很大。尚未有巴西患者携带该基因种系突变的报道。在本研究中,对表现出XP综合征主要临床特征的巴西患者进行了XPA种系突变状态的测定。该研究对来自选定巴西家庭的27名无亲缘关系的患者进行。仅在一名有神经功能障碍病史和轻度皮肤异常的患者中鉴定出双等位基因失活转换突变c.619C>T(p.Arg207Ter)。这些发现表明,在巴西人群中,XP综合征很少与遗传性致病XPA突变相关。此外,本报告证明了基因型-表型相关性作为指导直接基因筛查的有价值工具的有效性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/eb77/4425119/aa92bc505c77/ijms-16-08988-g001.jpg

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