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家族性心肌疾病相关 LMNA 基因突变的 iPSC 衍生心肌细胞的基因表达、结构和收缩性研究。

A Study of Gene Expression, Structure, and Contractility of iPSC-Derived Cardiac Myocytes from a Family with Heart Disease due to LMNA Mutation.

机构信息

Department of Biomedical Engineering, University of California, Irvine, CA, 92697, USA.

UCI Edwards Lifesciences Foundation Cardiovascular Innovation and Research Center (CIRC), University of California, Irvine, CA, 92697, USA.

出版信息

Ann Biomed Eng. 2021 Dec;49(12):3524-3539. doi: 10.1007/s10439-021-02850-8. Epub 2021 Sep 28.

DOI:10.1007/s10439-021-02850-8
PMID:34585335
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8671287/
Abstract

Genetic mutations to the Lamin A/C gene (LMNA) can cause heart disease, but the mechanisms making cardiac tissues uniquely vulnerable to the mutations remain largely unknown. Further, patients with LMNA mutations have highly variable presentation of heart disease progression and type. In vitro patient-specific experiments could provide a powerful platform for studying this phenomenon, but the use of induced pluripotent stem cell-derived cardiomyocytes (iPSC-CM) introduces heterogeneity in maturity and function thus complicating the interpretation of the results of any single experiment. We hypothesized that integrating single cell RNA sequencing (scRNA-seq) with analysis of the tissue architecture and contractile function would elucidate some of the probable mechanisms. To test this, we investigated five iPSC-CM lines, three controls and two patients with a (c.357-2A>G) mutation. The patient iPSC-CM tissues had significantly weaker stress generation potential than control iPSC-CM tissues demonstrating the viability of our in vitro approach. Through scRNA-seq, differentially expressed genes between control and patient lines were identified. Some of these genes, linked to quantitative structural and functional changes, were cardiac specific, explaining the targeted nature of the disease progression seen in patients. The results of this work demonstrate the utility of combining in vitro tools in exploring heart disease mechanics.

摘要

基因突变为 lamin A/C 基因(LMNA)可导致心脏病,但使心脏组织对突变特别脆弱的机制在很大程度上仍不清楚。此外,LMNA 突变的患者心脏病进展和类型的表现具有高度可变性。体外患者特异性实验可以为研究这种现象提供一个强大的平台,但诱导多能干细胞衍生的心肌细胞(iPSC-CM)的使用在成熟度和功能上引入了异质性,从而使任何单个实验的结果的解释复杂化。我们假设将单细胞 RNA 测序(scRNA-seq)与组织结构和收缩功能分析相结合,将阐明一些可能的机制。为了验证这一点,我们研究了五个 iPSC-CM 系,三个对照和两个具有(c.357-2A>G)突变的患者。与对照 iPSC-CM 组织相比,患者 iPSC-CM 组织的应激产生潜力明显较弱,证明了我们体外方法的可行性。通过 scRNA-seq,鉴定了对照和患者系之间差异表达的基因。其中一些与定量结构和功能变化相关的基因是心脏特异性的,解释了患者中观察到的疾病进展的靶向性质。这项工作的结果表明,结合体外工具探索心脏病力学的实用性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bfbf/8671287/233461d99505/10439_2021_2850_Fig5_HTML.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bfbf/8671287/233461d99505/10439_2021_2850_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bfbf/8671287/ed0e7e5c0e02/10439_2021_2850_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bfbf/8671287/692feb75dbff/10439_2021_2850_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bfbf/8671287/07aca2cf4744/10439_2021_2850_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bfbf/8671287/b5e0ec9d1950/10439_2021_2850_Fig4_HTML.jpg
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