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利用来自一名哈钦森-吉尔福德早衰综合征患者的诱导多能干细胞对心脏早衰进行建模。

Modelling premature cardiac aging with induced pluripotent stem cells from a hutchinson-gilford Progeria Syndrome patient.

作者信息

Monnerat Gustavo, Kasai-Brunswick Tais Hanae, Asensi Karina Dutra, Silva Dos Santos Danubia, Barbosa Raiana Andrade Quintanilha, Cristina Paccola Mesquita Fernanda, Calvancanti Albuquerque Joao Paulo, Raphaela Pires Ferreira, Wendt Camila, Miranda Kildare, Domont Gilberto Barbosa, Nogueira Fábio César Sousa, Bastos Carvalho Adriana, Campos de Carvalho Antonio Carlos

机构信息

Institute of Biophysics Carlos Chagas Filho, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.

Laboratory of Proteomics, LADETEC, Institute of Chemistry, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.

出版信息

Front Physiol. 2022 Nov 23;13:1007418. doi: 10.3389/fphys.2022.1007418. eCollection 2022.

Abstract

Hutchinson-Gilford Progeria Syndrome (HGPS) is a rare genetic disorder that causes accelerated aging and a high risk of cardiovascular complications. However, the underlying mechanisms of cardiac complications of this syndrome are not fully understood. This study modeled HGPS using cardiomyocytes (CM) derived from induced pluripotent stem cells (iPSC) derived from a patient with HGPS and characterized the biophysical, morphological, and molecular changes found in these CM compared to CM derived from a healthy donor. Electrophysiological recordings suggest that the HGPS-CM was functional and had normal electrophysiological properties. Electron tomography showed nuclear morphology alteration, and the 3D reconstruction of electron tomography images suggests structural abnormalities in HGPS-CM mitochondria, however, there was no difference in mitochondrial content as measured by Mitotracker. Immunofluorescence indicates nuclear morphological alteration and confirms the presence of Troponin T. Telomere length was measured using qRT-PCR, and no difference was found in the CM from HGPS when compared to the control. Proteomic analysis was carried out in a high-resolution system using Liquid Chromatography Tandem Mass Spectrometry (LC-MS/MS). The proteomics data show distinct group separations and protein expression differences between HGPS and control-CM, highlighting changes in ribosomal, TCA cycle, and amino acid biosynthesis, among other modifications. Our findings show that iPSC-derived cardiomyocytes from a Progeria Syndrome patient have significant changes in mitochondrial morphology and protein expression, implying novel mechanisms underlying premature cardiac aging.

摘要

哈钦森-吉尔福德早衰综合征(HGPS)是一种罕见的遗传性疾病,会导致加速衰老和心血管并发症的高风险。然而,该综合征心脏并发症的潜在机制尚未完全了解。本研究使用从一名HGPS患者诱导多能干细胞(iPSC)衍生而来的心肌细胞(CM)对HGPS进行建模,并将这些CM与来自健康供体的CM进行比较,以表征其生物物理、形态和分子变化。电生理记录表明,HGPS-CM功能正常,具有正常的电生理特性。电子断层扫描显示核形态改变,电子断层扫描图像的三维重建表明HGPS-CM线粒体存在结构异常,然而,用Mitotracker测量的线粒体含量没有差异。免疫荧光表明核形态改变,并证实了肌钙蛋白T的存在。使用qRT-PCR测量端粒长度,与对照组相比,HGPS的CM中未发现差异。使用液相色谱串联质谱(LC-MS/MS)在高分辨率系统中进行蛋白质组学分析。蛋白质组学数据显示HGPS和对照CM之间有明显的分组分离和蛋白质表达差异,突出了核糖体、三羧酸循环和氨基酸生物合成等方面的变化以及其他修饰。我们的研究结果表明,早衰综合征患者的iPSC衍生心肌细胞在线粒体形态和蛋白质表达方面有显著变化,这意味着心脏早衰的新机制。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ee2e/9726722/c1c832f94d7d/fphys-13-1007418-g001.jpg

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