Department of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, NY, USA.
Institute of Medical Genetics, University Hospital of Wales, Cardiff, UK.
Genet Med. 2019 Nov;21(11):2605-2613. doi: 10.1038/s41436-019-0537-7. Epub 2019 May 10.
With the advent of precision and genomic medicine, a critical issue is whether a disease gene variant is pathogenic or benign. Such is the case for the three autosomal dominant acute hepatic porphyrias (AHPs), including acute intermittent porphyria, hereditary coproporphyria, and variegate porphyria, each resulting from the half-normal enzymatic activities of hydroxymethylbilane synthase, coproporphyrinogen oxidase, and protoporphyrinogen oxidase, respectively. To date, there is no public database that documents the likely pathogenicity of variants causing the porphyrias, and more specifically, the AHPs with biochemically and clinically verified information. Therefore, an international collaborative with the European Porphyria Network and the National Institutes of Health/National Center for Advancing Translational Sciences/National Institute of Diabetes and Digestive and Kidney Diseases (NIH/NCATS/NIDDK)-sponsored Porphyrias Consortium of porphyria diagnostic experts is establishing an online database that will collate biochemical and clinical evidence verifying the pathogenicity of the published and newly identified variants in the AHP-causing genes. The overall goal of the International Porphyria Molecular Diagnostic Collaborative is to determine the pathogenic and benign variants for all eight porphyrias. Here we describe the overall objectives and the initial efforts to validate pathogenic and benign variants in the respective heme biosynthetic genes causing the AHPs.
随着精准医学和基因组医学的出现,一个关键问题是疾病基因变异是致病性的还是良性的。三种常染色体显性急性肝卟啉症(AHPs)就是这种情况,包括急性间歇性卟啉症、遗传性粪卟啉症和变异性卟啉症,每种疾病分别由羟甲基胆素合酶、粪卟啉原氧化酶和原卟啉原氧化酶的半正常酶活性引起。迄今为止,还没有公共数据库记录导致卟啉症的变异的可能致病性,更具体地说,还没有记录具有生物化学和临床验证信息的 AHP 的变异。因此,一个由欧洲卟啉症网络和美国国立卫生研究院/国家先进转化科学中心/国家糖尿病、消化和肾脏疾病研究所(NIH/NCATS/NIDDK)赞助的国际合作卟啉症诊断专家协会正在建立一个在线数据库,该数据库将整理生物化学和临床证据,以验证 AHP 致病基因中已发表和新鉴定的变异的致病性。国际卟啉症分子诊断协作组织的总体目标是确定所有八种卟啉症的致病性和良性变异。在这里,我们描述了验证导致 AHP 的血红素生物合成基因中致病性和良性变异的总体目标和初步努力。