Naya Francisco J, Black Brian L, Wu Hai, Bassel-Duby Rhonda, Richardson James A, Hill Joseph A, Olson Eric N
Department of Molecular Biology, University of Texas Southwestern Medical Center at Dallas, Dallas, Texas, USA.
Nat Med. 2002 Nov;8(11):1303-9. doi: 10.1038/nm789. Epub 2002 Oct 15.
The four MEF2 transcription factors (MEF2A, -B, -C, and -D) regulate differentiation and calcium-dependent gene expression in muscle cells. We generated mice deficient in MEF2A, the predominant Mef2 gene product expressed in post-natal cardiac muscle. Most mice lacking Mef2a died suddenly within the first week of life and exhibited pronounced dilation of the right ventricle, myofibrillar fragmentation, mitochondrial disorganization and activation of a fetal cardiac gene program. The few Mef2a(-/-) mice that survived to adulthood also showed a deficiency of cardiac mitochondria and susceptibility to sudden death. Paradoxically, MEF2 transcriptional activity, revealed by the expression of a MEF2-dependent transgene, was enhanced in the hearts of Mef2a-mutant mice, reflecting the transcriptional activation of residual MEF2D. These findings reveal specific roles for MEF2A in maintaining appropriate mitochondrial content and cyto-architectural integrity in the post-natal heart and show that other MEF2 isoforms cannot support these activities.
四种MEF2转录因子(MEF2A、-B、-C和-D)调节肌肉细胞的分化和钙依赖性基因表达。我们构建了MEF2A基因缺失的小鼠,MEF2A是出生后心肌中表达的主要Mef2基因产物。大多数缺乏Mef2a的小鼠在出生后第一周内突然死亡,并表现出右心室明显扩张、肌原纤维断裂、线粒体紊乱以及胎儿心脏基因程序的激活。少数存活至成年的Mef2a(-/-)小鼠也表现出心脏线粒体缺乏和猝死易感性。矛盾的是,由MEF2依赖性转基因表达所揭示的MEF2转录活性在Mef2a突变小鼠的心脏中增强,这反映了残余MEF2D的转录激活。这些发现揭示了MEF2A在维持出生后心脏中适当的线粒体含量和细胞结构完整性方面的特定作用,并表明其他MEF2亚型不能支持这些活动。