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在临床实践中进行法布里病的甲基组谱分析:概念验证。

Methylome Profiling in Fabry Disease in Clinical Practice: A Proof of Concept.

机构信息

CEINGE, Biotecnologie Avanzate, Via Gaetano Salvatore, 80145 Naples, Italy.

Department of Public Health, University Federico II, 80131 Naples, Italy.

出版信息

Int J Mol Sci. 2022 Oct 11;23(20):12110. doi: 10.3390/ijms232012110.

Abstract

Anderson−Fabry disease (FD) is an X-linked disease caused by a functional deficit of the α-galactosidase A enzyme. FD diagnosis relies on the clinical manifestations and research of GLA gene mutations. However, because of the lack of a clear genotype/phenotype correlation, FD diagnosis can be challenging. Recently, several studies have highlighted the importance of investigating DNA methylation patterns for confirming the correct diagnosis of different rare Mendelian diseases, but to date, no such studies have been reported for FD. Thus, in the present investigation, we analyzed for the first time the genome-wide methylation profile of a well-characterized cohort of patients with Fabry disease. We profiled the methylation status of about 850,000 CpG sites in 5 FD patients, all carrying the same mutation in the GLA gene (exon 6 c.901C>G) and presenting comparable low levels of α-Gal A activity. We found that, although the whole methylome profile did not discriminate the FD group from the unaffected one, several genes were significantly differentially methylated in Fabry patients. Thus, we provide here a proof of concept, to be tested in patients with different mutations and in a larger cohort, that the methylation state of specific genes can potentially identify Fabry patients and possibly predict organ involvement and disease evolution.

摘要

安德森-法布里病(FD)是一种 X 连锁疾病,由α-半乳糖苷酶 A 酶的功能缺陷引起。FD 的诊断依赖于临床表现和 GLA 基因突变的研究。然而,由于缺乏明确的基因型/表型相关性,FD 的诊断具有挑战性。最近,几项研究强调了研究 DNA 甲基化模式对于确认不同罕见孟德尔疾病正确诊断的重要性,但迄今为止,尚未有针对 FD 的此类研究报告。因此,在本研究中,我们首次分析了一组经过充分表征的法布里病患者的全基因组甲基化谱。我们对 5 名 FD 患者约 850,000 个 CpG 位点的甲基化状态进行了分析,所有患者的 GLA 基因(exon 6 c.901C>G)都携带相同的突变,并且α-Gal A 活性水平相当低。我们发现,尽管整个甲基化组图谱不能将 FD 组与未受影响的组区分开来,但在法布里病患者中,有几个基因的甲基化水平存在显著差异。因此,我们在这里提供了一个概念验证,有待在具有不同突变的患者和更大的队列中进行测试,即特定基因的甲基化状态可能能够识别法布里病患者,并可能预测器官受累和疾病进展。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fa8c/9602470/556f57c4135f/ijms-23-12110-g001.jpg

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