Robinson P N, Booms P
Institute of Medical Genetics, Department of General Pediatrics, Charité University Hospital, Berlin, Germany.
Cell Mol Life Sci. 2001 Oct;58(11):1698-707. doi: 10.1007/pl00000807.
The Marfan syndrome (MFS) is an autosomal dominant heritable disorder of connective tissue with highly variable clinical manifestations including aortic dilatation and dissection, ectopia lentis, and a range of skeletal anomalies. Mutations in the gene for fibrillin-1 (FBN1) cause MFS and other related disorders of connective tissue collectively termed type-1 fibrillinopathies. Fibrillin-1 is a main component of the 10- to 12-nm extracellular microfibrils that are important for elastogenesis, elasticity, and homeostasis of elastic fibers. Mutations in fibrillin-1 are hypothesized to exert their effects by dominant negative mechanisms, but recent work has also emphasized the potential role of proteases and disturbances in tissue homeostasis in the pathogenesis of the MFS. This article provides an overview of the clinical aspects of the MFS and current thinking on the pathogenesis of this disorder.
马凡综合征(MFS)是一种常染色体显性遗传性结缔组织疾病,临床表现高度可变,包括主动脉扩张和夹层、晶状体异位以及一系列骨骼异常。原纤维蛋白-1(FBN1)基因的突变导致MFS和其他相关的结缔组织疾病,统称为1型原纤维蛋白病。原纤维蛋白-1是10至12纳米细胞外微原纤维的主要成分,对弹性纤维的弹性生成、弹性和稳态至关重要。原纤维蛋白-1的突变被认为通过显性负性机制发挥作用,但最近的研究也强调了蛋白酶和组织稳态紊乱在MFS发病机制中的潜在作用。本文概述了MFS的临床方面以及目前对该疾病发病机制的认识。