• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

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

全球蛋白质组学分析揭示了病态层粘连蛋白 A/C 突变中差异表达蛋白的变化。

Global Proteomic Analysis Reveals Alterations in Differentially Expressed Proteins between Cardiopathic Lamin A/C Mutations.

机构信息

Department of Medicine, Division of Cardiovascular Medicine, University of Wisconsin-Madison, Madison, Wisconsin 53705, United States.

Department of Cell and Regenerative Biology, Human Proteomics Program, University of Wisconsin-Madison, Madison, Wisconsin 53705, United States.

出版信息

J Proteome Res. 2024 Jun 7;23(6):1970-1982. doi: 10.1021/acs.jproteome.3c00853. Epub 2024 May 8.

DOI:10.1021/acs.jproteome.3c00853
PMID:38718259
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11218822/
Abstract

Lamin A/C (LMNA) is an important component of nuclear lamina. Mutations cause arrhythmia, heart failure, and sudden cardiac death. While LMNA-associated cardiomyopathy typically has an aggressive course that responds poorly to conventional heart failure therapies, there is variability in severity and age of penetrance between and even within specific mutations, which is poorly understood at the cellular level. Further, this heterogeneity has not previously been captured to mimic the heterozygous state, nor have the hundreds of clinical LMNA mutations been represented. Herein, we have overexpressed cardiopathic LMNA variants in HEK cells and utilized state-of-the-art quantitative proteomics to compare the global proteomic profiles of (1) aggregating Q353 K alone, (2) Q353 K coexpressed with WT, (3) aggregating N195 K coexpressed with WT, and (4) nonaggregating E317 K coexpressed with WT to help capture some of the heterogeneity between mutations. We analyzed each data set to obtain the differentially expressed proteins (DEPs) and applied gene ontology (GO) and KEGG pathway analyses. We found a range of 162 to 324 DEPs from over 6000 total protein IDs with differences in GO terms, KEGG pathways, and DEPs important in cardiac function, further highlighting the complexity of cardiac laminopathies. Pathways disrupted by LMNA mutations were validated with redox, autophagy, and apoptosis functional assays in both HEK 293 cells and in induced pluripotent stem cell derived cardiomyocytes (iPSC-CMs) for LMNA N195 K. These proteomic profiles expand our repertoire for mutation-specific downstream cellular effects that may become useful as druggable targets for personalized medicine approach for cardiac laminopathies.

摘要

核纤层蛋白 A/C(LMNA)是核纤层的重要组成部分。突变会导致心律失常、心力衰竭和心源性猝死。虽然 LMNA 相关心肌病通常具有侵袭性病程,对传统心力衰竭治疗反应不佳,但特定突变之间甚至同一突变内的严重程度和发病年龄存在变异性,这在细胞水平上理解甚少。此外,这种异质性以前没有被捕获来模拟杂合状态,也没有代表数百种临床 LMNA 突变。在此,我们在 HEK 细胞中过表达了致病变异的 LMNA,并利用最先进的定量蛋白质组学技术,比较了(1)单独聚集的 Q353 K,(2)与 WT 共表达的 Q353 K,(3)与 WT 共表达的聚集的 N195 K,和(4)与 WT 共表达的非聚集的 E317 K 的全局蛋白质组谱,以帮助捕捉突变之间的一些异质性。我们分析了每个数据集以获得差异表达蛋白(DEPs),并应用基因本体论(GO)和 KEGG 途径分析。我们从超过 6000 个总蛋白质 ID 中获得了范围为 162 到 324 个 DEPs,GO 术语、KEGG 途径和对心脏功能重要的 DEPs 存在差异,进一步突出了心脏层粘连蛋白病的复杂性。通过在 HEK 293 细胞和诱导多能干细胞衍生的心肌细胞(iPSC-CMs)中针对 LMNA N195 K 进行的氧化还原、自噬和细胞凋亡功能测定,验证了由 LMNA 突变破坏的途径。这些蛋白质组谱扩展了我们对突变特异性下游细胞效应的了解,这可能成为心脏层粘连蛋白病个性化医学方法的有价值的药物靶点。

相似文献

1
Global Proteomic Analysis Reveals Alterations in Differentially Expressed Proteins between Cardiopathic Lamin A/C Mutations.全球蛋白质组学分析揭示了病态层粘连蛋白 A/C 突变中差异表达蛋白的变化。
J Proteome Res. 2024 Jun 7;23(6):1970-1982. doi: 10.1021/acs.jproteome.3c00853. Epub 2024 May 8.
2
-Related Dilated Cardiomyopathy: Single-Cell Transcriptomics during Patient-Derived iPSC Differentiation Support Cell Type and Lineage-Specific Dysregulation of Gene Expression and Development for Cardiomyocytes and Epicardium-Derived Cells with Lamin A/C Haploinsufficiency.相关扩张型心肌病:患者来源 iPSC 分化过程中的单细胞转录组学研究支持肌细胞和心外膜衍生细胞的基因表达和发育的细胞类型和谱系特异性失调,伴有核纤层蛋白 A/C 单倍不足。
Cells. 2024 Sep 3;13(17):1479. doi: 10.3390/cells13171479.
3
DNA Damage Response/TP53 Pathway Is Activated and Contributes to the Pathogenesis of Dilated Cardiomyopathy Associated With LMNA (Lamin A/C) Mutations.DNA 损伤反应/TP53 通路被激活并有助于伴有 LMNA(核纤层蛋白 A/C)突变的扩张型心肌病的发病机制。
Circ Res. 2019 Mar 15;124(6):856-873. doi: 10.1161/CIRCRESAHA.118.314238.
4
Gene expression profiling of fibroblasts in a family with LMNA-related cardiomyopathy reveals molecular pathways implicated in disease pathogenesis.家族性 LMNA 相关心肌病成纤维细胞的基因表达谱分析揭示了疾病发病机制中涉及的分子途径。
BMC Med Genet. 2020 Jul 22;21(1):152. doi: 10.1186/s12881-020-01088-w.
5
Dual specificity phosphatase 4 mediates cardiomyopathy caused by lamin A/C (LMNA) gene mutation.双重特异性磷酸酶 4 介导肌节蛋白 A/C(LMNA)基因突变引起的心肌病。
J Biol Chem. 2012 Nov 23;287(48):40513-24. doi: 10.1074/jbc.M112.404541. Epub 2012 Oct 9.
6
Functional Characterization of a Novel Truncating Mutation in Lamin A/C Gene in a Family with a Severe Cardiomyopathy with Conduction Defects.一个患有严重心肌病伴传导缺陷的家族中,核纤层蛋白A/C基因新的截短突变的功能特征分析
Cell Physiol Biochem. 2017;44(4):1559-1577. doi: 10.1159/000485651. Epub 2017 Dec 4.
7
From gene to mechanics: a comprehensive insight into the mechanobiology of LMNA mutations in cardiomyopathy.从基因到力学:肌原纤维核纤层蛋白 A 突变性心肌病的力学生物学综合分析
Cell Commun Signal. 2024 Mar 27;22(1):197. doi: 10.1186/s12964-024-01546-5.
8
The LMNA mutation p.Arg321Ter associated with dilated cardiomyopathy leads to reduced expression and a skewed ratio of lamin A and lamin C proteins.与扩张型心肌病相关的 LMNA 突变 p.Arg321Ter 导致 lamin A 和 lamin C 蛋白表达减少和比例失调。
Exp Cell Res. 2013 Nov 15;319(19):3010-9. doi: 10.1016/j.yexcr.2013.08.024. Epub 2013 Aug 31.
9
Modeling Treatment Response for Lamin A/C Related Dilated Cardiomyopathy in Human Induced Pluripotent Stem Cells.人类诱导多能干细胞中与核纤层蛋白A/C相关的扩张型心肌病的治疗反应建模
J Am Heart Assoc. 2017 Jul 28;6(8):e005677. doi: 10.1161/JAHA.117.005677.
10
Temsirolimus activates autophagy and ameliorates cardiomyopathy caused by lamin A/C gene mutation.他莫昔芬激活自噬并改善由 lamin A/C 基因突变引起的心肌病。
Sci Transl Med. 2012 Jul 25;4(144):144ra102. doi: 10.1126/scitranslmed.3003875.

引用本文的文献

1
Coordination of Focal Adhesion Nanoarchitecture and Dynamics in Mechanosensing for Cardiomyoblast Differentiation.心肌成纤维细胞分化机械传感中粘着斑纳米结构与动力学的协调
ACS Appl Mater Interfaces. 2025 Jan 22;17(3):4463-4479. doi: 10.1021/acsami.4c15459. Epub 2025 Jan 8.

本文引用的文献

1
Nuclear proteostasis imbalance in laminopathy-associated premature aging diseases.核蛋白稳态失衡与层粘连蛋白病相关的早老性疾病。
FASEB J. 2023 Aug;37(8):e23116. doi: 10.1096/fj.202300878R.
2
Aberrant chromatin organization at the nexus of laminopathy disease pathways.核纤层蛋白病疾病途径交叉点处的异常染色质组织。
Nucleus. 2022 Dec;13(1):300-312. doi: 10.1080/19491034.2022.2153564.
3
ER stress and calcium-dependent arrhythmias.内质网应激与钙依赖性心律失常。
Front Physiol. 2022 Nov 8;13:1041940. doi: 10.3389/fphys.2022.1041940. eCollection 2022.
4
Biallelic loss of LDB3 leads to a lethal pediatric dilated cardiomyopathy.LDB3 双等位基因缺失导致致命性小儿扩张型心肌病。
Eur J Hum Genet. 2023 Jan;31(1):97-104. doi: 10.1038/s41431-022-01204-9. Epub 2022 Oct 17.
5
Lamin A/C impairments cause mitochondrial dysfunction by attenuating PGC1α and the NAMPT-NAD+ pathway.核纤层蛋白 A/C 缺陷通过减弱 PGC1α 和 NAMPT-NAD+ 通路引起线粒体功能障碍。
Nucleic Acids Res. 2022 Sep 23;50(17):9948-9965. doi: 10.1093/nar/gkac741.
6
BNIP-2 Activation of Cellular Contractility Inactivates YAP for H9c2 Cardiomyoblast Differentiation.BNIP-2 通过激活细胞收缩性使 YAP 在 H9c2 心肌细胞分化中失活。
Adv Sci (Weinh). 2022 Nov;9(31):e2202834. doi: 10.1002/advs.202202834. Epub 2022 Aug 17.
7
dia-PASEF data analysis using FragPipe and DIA-NN for deep proteomics of low sample amounts.使用 FragPipe 和 DIA-NN 对低样本量进行深度蛋白质组学分析的 dia-PASEF 数据分析。
Nat Commun. 2022 Jul 8;13(1):3944. doi: 10.1038/s41467-022-31492-0.
8
Deficiency of ribosomal proteins reshapes the transcriptional and translational landscape in human cells.核糖体蛋白缺失重塑了人类细胞的转录和翻译景观。
Nucleic Acids Res. 2022 Jul 8;50(12):6601-6617. doi: 10.1093/nar/gkac053.
9
Phenotypic Variability in iPSC-Induced Cardiomyocytes and Cardiac Fibroblasts Carrying Diverse Mutations.携带不同突变的诱导多能干细胞衍生心肌细胞和心脏成纤维细胞的表型变异性。
Front Physiol. 2021 Dec 16;12:778982. doi: 10.3389/fphys.2021.778982. eCollection 2021.
10
Modulation of muscle redox and protein aggregation rescues lethality caused by mutant lamins.调节肌肉氧化还原和蛋白质聚集可挽救由突变型核纤层蛋白引起的致死性。
Redox Biol. 2021 Nov 25;48:102196. doi: 10.1016/j.redox.2021.102196.