Suppr超能文献

散发性迟发性皮肤卟啉症(sPCT)中的血色素沉着症(HFE)和转铁蛋白受体-1(TFRC1)基因。

Hemochromatosis (HFE) and transferrin receptor-1 (TFRC1) genes in sporadic porphyria cutanea tarda (sPCT).

作者信息

Lamoril Jérĵme, Andant Christophe, Gouya Laurent, Malonova Eva, Grandchamp Bernard, Martásek Pavel, Deybac Jean-Charles, Puy Hervé

机构信息

Centre Français des Porphyries, INSERM U409, Service de Biochimie, Hĵpital Louis Mourier, Colombes, France.

出版信息

Cell Mol Biol (Noisy-le-grand). 2002 Feb;48(1):33-41.

Abstract

Porphyria cutanea tarda (PCT), a disorder characterized by a photosensitive dermatosis and hepatic siderosis, is caused by a decreased activity of the hepatic enzyme uroporphyrinogen decarboxylase (UROD). Two forms of PCT have been described: a familial one (fPCT) with an inherited decrease of UROD activity in all tissues and a sporadic one (sPCT) with a decreased UROD activity restricted to the liver. Iron overload and acquired factors including hepatic viral infections, alcohol, drugs contribute to the expression of PCT. In 65 French sPCT patients and 108 controls we have evaluated the respective role of iron and HCV status, the hemochromatosis (HFE) gene mutations frequencies (H63D. S65C, C282Y), and in a case control study we searched for an association between sPCT and the human transferrin receptor-1 (TFRC1) gene whose product is thought to be in functional association with the HFE protein: three single nucleotide polymorphisms (SNPs) previously characterized and 2 novel ones were studied. The iron-related parameters and transaminases were higher in sPCT patients than those of non-porphyric controls. Of the sPCT patients studied, 28% were HCV positive. In the HFE gene, 17% of sPCT patients carried C282Y mutation compared to 4% in controls, no significant differences were found with H63D and S65C variants. Compound heterozygous genotypes, C282Y/H63D or C282Y/S65C, were not significantly different in sPCT and control groups. Independently from HFE gene mutations, an association was found between the IVS4+198 T allele in the TFRC1 gene and sPCT patients. Analysis of HFE genotypes indicated that C282Y (but not H63D nor S65C) is a susceptibility factor for the development of sPCT in West European continental patients. However, analysis of TFRC1 genotypes suggest that sPCT should be considered as a multifactorial disorder in which other intracellular iron metabolism genes could be involved.

摘要

迟发性皮肤卟啉症(PCT)是一种以光敏性皮肤病和肝铁质沉着为特征的疾病,由肝酶尿卟啉原脱羧酶(UROD)活性降低引起。PCT有两种类型:一种是家族性的(fPCT),所有组织中UROD活性遗传性降低;另一种是散发性的(sPCT),UROD活性降低仅限于肝脏。铁过载和包括肝病毒感染、酒精、药物在内的后天因素促成了PCT的表现。在65名法国sPCT患者和108名对照中,我们评估了铁和丙型肝炎病毒(HCV)状态的各自作用、血色素沉着症(HFE)基因突变频率(H63D、S65C、C282Y),并且在一项病例对照研究中,我们寻找sPCT与人类转铁蛋白受体-1(TFRC1)基因之间的关联,该基因的产物被认为与HFE蛋白存在功能关联:研究了先前鉴定的三个单核苷酸多态性(SNP)和两个新的SNP。sPCT患者的铁相关参数和转氨酶高于非卟啉症对照。在所研究的sPCT患者中,28%为HCV阳性。在HFE基因中,17%的sPCT患者携带C282Y突变,而对照中为4%,H63D和S65C变体未发现显著差异。复合杂合基因型C282Y/H63D或C282Y/S65C在sPCT组和对照组中无显著差异。独立于HFE基因突变,在TFRC1基因的IVS4+198 T等位基因与sPCT患者之间发现了关联。HFE基因型分析表明,C282Y(而非H63D或S65C)是西欧大陆患者发生sPCT的易感因素。然而,TFRC1基因型分析表明,sPCT应被视为一种多因素疾病,其中可能涉及其他细胞内铁代谢基因。

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验