Department of Cardiology, Thorax Centre, University Medical Center Groningen, University of Groningen, Groningen, the Netherlands.
Neth Heart J. 2010 Feb;18(2):85-9. doi: 10.1007/BF03091743.
Background/Methods. Marfan syndrome (MFS) is a heritable connective tissue disorder usually caused by a mutation in the fibrillin 1 (FBN1) gene. Typical characteristics of MFS that have been described include dolichostenomelia, ectopia lentis and aortic root dilatation. However, there is great clinical variability in the expression of the syndrome's manifestations, both between and within families. Here we discuss the clinical variability of MFS by describing a large fourgeneration Dutch family with MFS.Results. Nineteen individuals of one family with a single missense FBN1 mutation (c.7916A>G) were identified. The same mutation was found in one unrelated person. Clinical variability was extensive and not all mutation carriers fulfilled the diagnostic criteria for MFS. Some patients only expressed mild skeletal abnormalities, whereas aortic root dilation was present in eight patients, an acute type A aortic dissection was recorded in two other patients, and a mitral valve prolapse was present in eight patients. In some patients cardiac features were not present on initial screening, but did however develop over time.Conclusion. MFS is a clinically highly variable syndrome, which means a meticulous evaluation of suspected cases is crucial. Mutation carriers should be re-evaluated regularly as cardiovascular symptoms may develop over time. (Neth Heart J 2010;18:85-9.).
背景/方法:马凡综合征(MFS)是一种遗传性结缔组织疾病,通常由原纤维蛋白 1(FBN1)基因的突变引起。MFS 的典型特征包括长骨细长、晶状体异位和主动脉根部扩张。然而,该综合征的临床表现存在很大的临床变异性,无论是在个体之间还是在家族内。在这里,我们通过描述一个具有 MFS 的大型四代荷兰家族来讨论 MFS 的临床变异性。
确定了一个家族中有 19 名个体携带单一错义 FBN1 突变(c.7916A>G)。在一个无关的人中也发现了相同的突变。临床变异性很广泛,并非所有突变携带者都符合 MFS 的诊断标准。一些患者仅表现出轻微的骨骼异常,而 8 名患者存在主动脉根部扩张,2 名其他患者出现急性 A 型主动脉夹层,8 名患者存在二尖瓣脱垂。在一些患者中,心脏特征在初始筛查时不存在,但随着时间的推移会出现。
MFS 是一种临床表现高度可变的综合征,这意味着对疑似病例进行仔细评估至关重要。突变携带者应定期重新评估,因为心血管症状可能会随着时间的推移而发展。(Neth Heart J 2010;18:85-9.)。