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法布里病人类足细胞系的全基因组表达分析。

Genome-wide expression analysis in a Fabry disease human podocyte cell line.

作者信息

Snanoudj Sarah, Derambure Céline, Zhang Cheng, Hai Yen Nguyen Thi, Lesueur Céline, Coutant Sophie, Abily-Donval Lénaïg, Marret Stéphane, Yang Hong, Mardinoglu Adil, Bekri Soumeya, Tebani Abdellah

机构信息

Normandie Univ, UNIROUEN, INSERM, U1245, CHU Rouen, Department of Metabolic Biochemistry, Referral Center for Lysosomal Diseases, Filière G2M, 76000, Rouen, France.

Normandie Univ, UNIROUEN, INSERM U1245 and CHU Rouen, Department of Genetics and Reference Center for Developmental Disorders, FHU-G4 Génomique, F-76000, Rouen, France.

出版信息

Heliyon. 2024 Jul 9;10(14):e34357. doi: 10.1016/j.heliyon.2024.e34357. eCollection 2024 Jul 30.

Abstract

Fabry disease (FD) is an X-linked lysosomal disease caused by an enzyme deficiency of alpha-galactosidase A (α-gal A). This deficiency leads to the accumulation of glycosphingolipids in lysosomes, resulting in a range of clinical symptoms. The complex pathogenesis of FD involves lysosomal dysfunction, altered autophagy, and mitochondrial abnormalities. Omics sciences, particularly transcriptomic analysis, comprehensively understand molecular mechanisms underlying diseases. This study focuses on genome-wide expression analysis in an FD human podocyte model to gain insights into the underlying mechanisms of podocyte dysfunction. Human control and GLA-edited podocytes were used. Gene expression data was generated using RNA-seq analysis, and differentially expressed genes were identified using DESeq2. Principal component analysis and Spearman correlation have explored gene expression trends. Functional enrichment and Reporter metabolite analyses were conducted to identify significantly affected metabolites and metabolic pathways. Differential expression analysis revealed 247 genes with altered expression levels in GLA-edited podocytes compared to control podocytes. Among these genes, 136 were underexpressed, and 111 were overexpressed in GLA-edited cells. Functional analysis of differentially expressed genes showed their involvement in various pathways related to oxidative stress, inflammation, fatty acid metabolism, collagen and extracellular matrix homeostasis, kidney injury, apoptosis, autophagy, and cellular stress response. The study provides insights into molecular mechanisms underlying Fabry podocyte dysfunction. Integrating transcriptomics data with genome-scale metabolic modeling further unveiled metabolic alterations in GLA-edited podocytes. This comprehensive approach contributes to a better understanding of Fabry disease and may lead to identifying new biomarkers and therapeutic targets for this rare lysosomal disorder.

摘要

法布里病(FD)是一种X连锁溶酶体疾病,由α-半乳糖苷酶A(α-gal A)的酶缺乏引起。这种缺乏导致糖鞘脂在溶酶体中积累,从而产生一系列临床症状。FD复杂的发病机制涉及溶酶体功能障碍、自噬改变和线粒体异常。组学科学,特别是转录组分析,有助于全面了解疾病背后的分子机制。本研究聚焦于FD人足细胞模型中的全基因组表达分析,以深入了解足细胞功能障碍的潜在机制。使用了人类对照和GLA编辑的足细胞。通过RNA测序分析生成基因表达数据,并使用DESeq2鉴定差异表达基因。主成分分析和斯皮尔曼相关性分析探索了基因表达趋势。进行了功能富集和报告代谢物分析,以识别受显著影响的代谢物和代谢途径。差异表达分析显示,与对照足细胞相比,GLA编辑的足细胞中有247个基因的表达水平发生了改变。在这些基因中,136个基因表达下调,111个基因在GLA编辑的细胞中表达上调。对差异表达基因的功能分析表明,它们参与了与氧化应激、炎症、脂肪酸代谢、胶原蛋白和细胞外基质稳态、肾损伤、细胞凋亡、自噬和细胞应激反应相关的各种途径。该研究深入了解了法布里足细胞功能障碍背后的分子机制。将转录组学数据与基因组规模代谢模型相结合,进一步揭示了GLA编辑的足细胞中的代谢改变。这种综合方法有助于更好地理解法布里病,并可能有助于识别这种罕见溶酶体疾病的新生物标志物和治疗靶点。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e82/11295972/8cb506a30a76/gr1.jpg

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