Ramachandra Chrishan J A, Mehta Ashish, Guo Kenneth Wei Qiang, Wong Philip, Tan Ju Le, Shim Winston
National Heart Research Institute Singapore, Singapore.
Department of Cardiology, National Heart Centre Singapore, Singapore.
Int J Cardiol. 2015;187:585-91. doi: 10.1016/j.ijcard.2015.03.423. Epub 2015 Mar 31.
Marfan syndrome (MFS) is a genetic disorder that affects multiple organs. Mortality imposed by aortic aneurysm and dissections represent the most serious clinical manifestation of MFS. Progressive pathological aortic root enlargement as the result of degeneration of microfibril architecture and consequential loss of extracellular matrix integrity due to fibrillin-1 (FBN1) mutations are commonly diagnosed clinical manifestations of MFS. However, overlapping clinical manifestations with other aneurysmal disorders present a significant challenge in early and accurate diagnosis of MFS. While FBN1 mutations, abnormal transforming growth factor-β signaling and dysregulated matrix metalloproteinases have been implicated in MFS, clinically accepted risk-stratifying biomarkers have yet to be reliably identified. In this review, we summarize current consensus and recent insights in the understanding of MFS pathogenesis. Finally, we introduce the application of induced pluripotent stem cells (iPSCs) as cellular models for MFS and its potential as a novel platform into providing better appreciation of mechanisms underlying MFS diverse manifestations in the cardiovascular system.
马凡综合征(MFS)是一种影响多个器官的遗传性疾病。主动脉瘤和夹层导致的死亡率是MFS最严重的临床表现。由于微原纤维结构退化以及因原纤蛋白-1(FBN1)突变导致细胞外基质完整性随之丧失,进而出现进行性病理性主动脉根部扩大,这是MFS常见的临床表现。然而,与其他动脉瘤性疾病重叠的临床表现给MFS的早期准确诊断带来了重大挑战。虽然FBN1突变、异常的转化生长因子-β信号传导和失调的基质金属蛋白酶与MFS有关,但尚未可靠地鉴定出临床上可接受的风险分层生物标志物。在这篇综述中,我们总结了目前对MFS发病机制的共识和最新见解。最后,我们介绍了诱导多能干细胞(iPSC)作为MFS细胞模型的应用及其作为一个新平台的潜力,以便更好地理解MFS在心血管系统中各种表现的潜在机制。