LMNA cardiomyopathy: cell biology and genetics meet clinical medicine

被引:80
作者
Lu, Jonathan T. [1 ]
Muchir, Antoine [1 ,2 ]
Nagy, Peter L. [2 ]
Worman, Howard J. [1 ,2 ]
机构
[1] Columbia Univ, Dept Med, Coll Phys & Surg, New York, NY 10032 USA
[2] Columbia Univ, Dept Pathol & Cell Biol, Coll Phys & Surg, New York, NY 10032 USA
基金
美国国家卫生研究院;
关键词
LAMIN A/C GENE; DILATED CARDIOMYOPATHY; NUCLEAR-ENVELOPE; MUSCULAR-DYSTROPHY; STRUCTURAL ORGANIZATION; MOUSE MODELS; MUTATIONS; IDENTIFICATION; INHIBITION; ACTIVATION;
D O I
10.1242/dmm.006346
中图分类号
Q2 [细胞生物学];
学科分类号
071009 ; 090102 ;
摘要
Mutations in the LMNA gene, which encodes A-type nuclear lamins (intermediate filament proteins expressed in most differentiated somatic cells), cause a diverse range of diseases, called laminopathies, that selectively affect different tissues and organ systems. The most prevalent laminopathy is cardiomyopathy with or without different types of skeletal muscular dystrophy. LMNA cardiomyopathy has an aggressive clinical course with higher rates of deadly arrhythmias and heart failure than most other heart diseases. As awareness among physicians increases, and advances in DNA sequencing methods make the genetic diagnosis of LMNA cardiomyopathy more common, cardiologists are being faced with difficult questions regarding patient management. These questions concern the optimal use of intracardiac cardioverter defibrillators to prevent sudden death from arrhythmias, and medical interventions to prevent heart damage and ameliorate heart failure symptoms. Data from a mouse model of LMNA cardiomyopathy suggest that inhibitors of mitogen-activated protein kinase (MAPK) signaling pathways are beneficial in preventing and treating cardiac dysfunction; this basic research discovery needs to be translated to human patients.
引用
收藏
页码:562 / 568
页数:7
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