Furthmayr H, Francke U
Department of Pathology, Stanford University Medical School, CA 94304-5324, USA.
Semin Thorac Cardiovasc Surg. 1997 Jul;9(3):191-205.
More than 70 unique fibrillin-1 mutations have been identified in individuals with a variety of phenotypic changes. These range from severe neonatal lethal forms of Marfan syndrome to adult onset manifestations, mitral valve prolapse syndromes to isolated features such as ectopia lentis, Marfanoid body habitus and ascending aortic aneurysm and/or dissection. Fibrillin-1 mutations result in structurally and functionally defective fibrillin-1 molecules and microfibrils. Recent molecular genetic and fibrillin-1 biosynthesis studies suggest that individuals with fibrillin-1 abnormalities can be further subdivided into groups that are associated with distinct differences in severity and prognosis. In recognition of the expanding scope of related connective tissue disorders, we propose the terms microfibrillar disorder for disorders affecting fibrillin-containing microfibrils, and the more narrow concept of fibrillinopathy for clinical entities associated with abnormalities of fibrillin-1 or fibrillin-2. This latter category includes the previously defined disorders Marfan syndrome, congenital contractual arachnodactyly, and forms of ascending aortic aneurysm and/or dissection.
在患有各种表型变化的个体中已鉴定出70多种独特的原纤维蛋白-1突变。这些变化范围从马凡综合征的严重新生儿致死形式到成人发病表现,从二尖瓣脱垂综合征到孤立特征,如晶状体异位、类马凡体型和升主动脉瘤和/或夹层。原纤维蛋白-1突变导致结构和功能有缺陷的原纤维蛋白-1分子和微原纤维。最近的分子遗传学和原纤维蛋白-1生物合成研究表明,原纤维蛋白-1异常的个体可进一步细分为与严重程度和预后存在明显差异相关的组。认识到相关结缔组织疾病范围的不断扩大,我们提出“微原纤维疾病”这一术语来指代影响含原纤维蛋白微原纤维的疾病,以及“原纤维蛋白病”这一更狭义的概念来指代与原纤维蛋白-1或原纤维蛋白-2异常相关的临床实体。后一类包括先前定义的疾病马凡综合征、先天性挛缩性蜘蛛指(趾)症以及升主动脉瘤和/或夹层的形式。