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弹性纤维碎片化的研究进展:马凡综合征主动脉瘤的发病机制与治疗。

Insights into elastic fiber fragmentation: Mechanisms and treatment of aortic aneurysm in Marfan syndrome.

机构信息

Department of Internal Medicine, Wayne State University, Detroit, MI, USA.

Center for Molecular Medicine and Genetics, Wayne State University, Detroit, MI, USA.

出版信息

Vascul Pharmacol. 2023 Dec;153:107215. doi: 10.1016/j.vph.2023.107215. Epub 2023 Aug 26.

Abstract

Marfan syndrome (MFS) is an autosomal dominant connective tissue disorder caused by mutations in fibrillin 1 (FBN1) gene. These mutations result in defects in the skeletal, ocular, and cardiovascular systems. Aortic aneurysm is the leading cause of premature mortality in untreated MFS patients. Elastic fiber fragmentation in the aortic vessel wall is a hallmark of MFS-associated aortic aneurysms. FBN1 mutations result in FBN1 fragments that also contribute to elastic fiber fragmentation. Although recent research has advanced our understanding of MFS, the contribution of elastic fiber fragmentation to the pathogenesis of aneurysm formation remains poorly understood. This review provides a comprehensive overview of the molecular mechanisms of elastic fiber fragmentation and its role in the pathogenesis of aortic aneurysm progression. Increased comprehension of elastic fragmentation has significant clinical implications for developing targeted interventions to block aneurysm progression, which would benefit not only individuals with Marfan syndrome but also other patients with aneurysms. Moreover, this review highlights an overlooked connection between inhibiting aneurysm and the restoration of elastic fibers in the vessel wall with various aneurysm inhibitors, including drugs and chemicals. Investigating the underlying molecular mechanisms could uncover innovative therapeutic strategies to inhibit elastin fragmentation and prevent the progression of aneurysms.

摘要

马凡综合征(MFS)是一种常染色体显性结缔组织疾病,由原纤维蛋白 1(FBN1)基因突变引起。这些突变导致骨骼、眼部和心血管系统的缺陷。主动脉瘤是未经治疗的 MFS 患者过早死亡的主要原因。主动脉血管壁中的弹性纤维断裂是 MFS 相关主动脉瘤的标志。FBN1 突变导致 FBN1 片段,也有助于弹性纤维断裂。尽管最近的研究提高了我们对 MFS 的理解,但弹性纤维断裂对动脉瘤形成发病机制的贡献仍知之甚少。这篇综述全面概述了弹性纤维断裂的分子机制及其在主动脉瘤进展发病机制中的作用。对弹性纤维断裂的理解的提高对开发靶向干预措施以阻止动脉瘤进展具有重要的临床意义,这不仅有益于马凡综合征患者,也有益于其他动脉瘤患者。此外,该综述强调了各种动脉瘤抑制剂(包括药物和化学物质)在抑制动脉瘤和恢复血管壁弹性纤维方面被忽视的联系。研究潜在的分子机制可以揭示抑制弹性蛋白断裂和防止动脉瘤进展的创新治疗策略。

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