McDermott-Roe Chris, Leleu Marion, Rowe Glenn C, Palygin Oleg, Bukowy John D, Kuo Judy, Rech Monika, Hermans-Beijnsberger Steffie, Schaefer Sebastian, Adami Eleonora, Creemers Esther E, Heinig Matthias, Schroen Blanche, Arany Zoltan, Petretto Enrico, Geurts Aron M
Cardiovascular Center, Medical College of Wisconsin, Milwaukee, WI, United States of America.
School of Life Sciences, Ecole Polytechnique Fédérale de Lausanne (EPFL), Lausanne, Switzerland.
PLoS One. 2017 Feb 3;12(2):e0170458. doi: 10.1371/journal.pone.0170458. eCollection 2017.
Mitochondrial dysfunction contributes to myriad monogenic and complex pathologies. To understand the underlying mechanisms, it is essential to define the full complement of proteins that modulate mitochondrial function. To identify such proteins, we performed a meta-analysis of publicly available gene expression data. Gene co-expression analysis of a large and heterogeneous compendium of microarray data nominated a sub-population of transcripts that whilst highly correlated with known mitochondrial protein-encoding transcripts (MPETs), are not themselves recognized as generating proteins either localized to the mitochondrion or pertinent to functions therein. To focus the analysis on a medically-important condition with a strong yet incompletely understood mitochondrial component, candidates were cross-referenced with an MPET-enriched module independently generated via genome-wide co-expression network analysis of a human heart failure gene expression dataset. The strongest uncharacterized candidate in the analysis was Leucine Rich Repeat Containing 2 (LRRC2). LRRC2 was found to be localized to the mitochondria in human cells and transcriptionally-regulated by the mitochondrial master regulator Pgc-1α. We report that Lrrc2 transcript abundance correlates with that of β-MHC, a canonical marker of cardiac hypertrophy in humans and experimentally demonstrated an elevation in Lrrc2 transcript in in vitro and in vivo rodent models of cardiac hypertrophy as well as in patients with dilated cardiomyopathy. RNAi-mediated Lrrc2 knockdown in a rat-derived cardiomyocyte cell line resulted in enhanced expression of canonical hypertrophic biomarkers as well as increased mitochondrial mass in the context of increased Pgc-1α expression. In conclusion, our meta-analysis represents a simple yet powerful springboard for the nomination of putative mitochondrially-pertinent proteins relevant to cardiac function and enabled the identification of LRRC2 as a novel mitochondrially-relevant protein and regulator of the hypertrophic response.
线粒体功能障碍会导致众多单基因和复杂疾病。为了解其潜在机制,明确调节线粒体功能的完整蛋白质组至关重要。为鉴定此类蛋白质,我们对公开可用的基因表达数据进行了荟萃分析。对大量异质性微阵列数据进行基因共表达分析,确定了一组转录本亚群,这些转录本虽与已知线粒体蛋白编码转录本(MPETs)高度相关,但本身并不被认为能产生定位于线粒体或与线粒体功能相关的蛋白质。为将分析聚焦于一种线粒体成分显著但尚未完全了解的医学重要病症,我们将候选基因与通过对人类心力衰竭基因表达数据集进行全基因组共表达网络分析独立生成的富含MPET的模块进行了交叉比对。分析中最突出的未表征候选基因是富含亮氨酸重复序列2(LRRC2)。我们发现LRRC2在人类细胞中定位于线粒体,且受线粒体主调节因子Pgc-1α转录调控。我们报告称,Lrrc2转录本丰度与β-MHC相关,β-MHC是人类心脏肥大的典型标志物,并且在体外和体内啮齿动物心脏肥大模型以及扩张型心肌病患者中,实验证明Lrrc2转录本升高。在大鼠来源的心肌细胞系中,RNAi介导的Lrrc2敲低导致典型肥大生物标志物表达增强,以及在Pgc-1α表达增加的情况下线粒体质量增加。总之,我们的荟萃分析为提名与心脏功能相关的假定线粒体相关蛋白提供了一个简单而强大的跳板,并使我们能够鉴定LRRC2为一种新型线粒体相关蛋白和肥大反应的调节因子。