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马凡综合征及相关疾病中的二尖瓣疾病。

Mitral valve disease in Marfan syndrome and related disorders.

机构信息

Division of Cardiology, Johns Hopkins University, Ross 1049 720 Rutland Avenue, Baltimore, MD 21205, USA.

出版信息

J Cardiovasc Transl Res. 2011 Dec;4(6):741-7. doi: 10.1007/s12265-011-9314-y. Epub 2011 Aug 25.

Abstract

Marfan syndrome (MFS) is a systemic disorder of the connective tissue with pleiotropic manifestations due to heterozygous FBN1 mutations and consequent upregulation of TGFβ signaling in affected tissues. Myxomatous thickening and elongation of the mitral valve (MV) leaflets commonly occur in this condition. Investigation of murine models of this disease has led to improved understanding of the mechanisms that underlie many of the phenotypic features of MFS, including MV disease. Loeys-Dietz syndrome (LDS) is a related disorder due to heterozygous mutations in the genes encoding subunits of the TGFβ receptor, and it may also involve the MV leaflets with similar elongation and thickening of the MV leaflets. Although the genetic basis and pathogenesis of nonsyndromic MV prolapse has been elusive to date, insights derived from monogenic disorders like MFS and LDS can be informative with regard to novel gene discovery and investigation into the pathogenesis of MV disease. This manuscript will review the prevalence of MV disease in MFS, its pathogenic basis as determined in mice with Fbn1 mutations, and ongoing studies that seek to better understand MV disease in the context of fibrillin-1 deficiency or excessive TGFβ signaling.

摘要

马凡综合征(MFS)是一种结缔组织的全身性疾病,由于 FBN1 突变的杂合性和随后受影响组织中 TGFβ 信号的上调,表现出多种表现。二尖瓣(MV)瓣叶的黏液瘤样增厚和伸长在这种情况下很常见。对这种疾病的小鼠模型的研究导致了对许多 MFS 表型特征(包括 MV 疾病)的潜在机制的更好理解。Loeys-Dietz 综合征(LDS)是一种相关疾病,由于编码 TGFβ 受体亚基的基因突变,也可能涉及 MV 瓣叶,MV 瓣叶也会出现类似的伸长和增厚。尽管迄今为止,非综合征性 MV 脱垂的遗传基础和发病机制仍难以捉摸,但从 MFS 和 LDS 等单基因疾病中获得的见解可以为新基因的发现和 MV 疾病发病机制的研究提供信息。本文将回顾 MFS 中 MV 疾病的患病率、在 Fbn1 突变小鼠中确定的其发病基础,以及正在进行的旨在更好地了解纤维连接蛋白 1 缺乏或 TGFβ 信号过度时 MV 疾病的研究。

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