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携带FBN1基因突变的西班牙裔家庭中骨骼表现稀少。

Paucity of skeletal manifestations in Hispanic families with FBN1 mutations.

作者信息

Villamizar Carlos, Regalado Ellen S, Fadulu Van Tran, Hasham Sumera N, Gupta Prateek, Willing Marcia C, Kuang Shao-Qing, Guo Dongchuan, Muilenburg Ann, Yee Richard W, Fan Yuxin, Towbin Jeffrey, Coselli Joseph S, LeMaire Scott A, Milewicz Dianna M

机构信息

Department of Internal Medicine, University of Texas Health Science Center at Houston, Houston, TX, USA.

出版信息

Eur J Med Genet. 2010 Mar-Apr;53(2):80-4. doi: 10.1016/j.ejmg.2009.11.001. Epub 2009 Nov 23.

Abstract

Marfan syndrome (MFS) is an autosomal dominant condition with pleiotropic manifestations involving the skeletal, ocular, and cardiovascular systems. The diagnosis is based primarily on clinical involvement of these and other systems, referred to as the Ghent criteria. We have identified three Hispanic families from Mexico with cardiovascular and ocular manifestations due to novel FBN1 mutations but with paucity of skeletal features. The largest family, hMFS001, had a frameshift mutation in exon 24 (3075delC) identified as the cause of aortic disease in the family. Assessment of eight affected adults revealed no major skeletal manifestation of MFS. Family hMFS002 had a missense mutation (R1530C) in exon 37. Four members fulfilled the criteria for ocular and cardiovascular phenotype but lacked skeletal manifestations. Family hMFS003 had two consecutive missense FBN1 mutations (C515W and R516G) in exon 12. Eight members fulfilled the ocular criteria for MFS and two members had major cardiovascular manifestations, however none of them met criteria for skeletal system. These data suggest that individuals of Hispanic descent with FBN1 mutations may not manifest skeletal features of the MFS to the same extent as Caucasians. We recommend that echocardiogram, ocular examination and FBN1 molecular testing be considered for any patients with possible MFS even in the absence of skeletal features, including Hispanic patients.

摘要

马凡综合征(MFS)是一种常染色体显性遗传病,具有多效性表现,累及骨骼、眼睛和心血管系统。诊断主要基于这些系统及其他系统的临床受累情况,即根特标准。我们从墨西哥确定了三个西班牙裔家族,这些家族因新的FBN1突变而出现心血管和眼部表现,但骨骼特征较少。最大的家族hMFS001在外显子24(3075delC)有一个移码突变,被确定为该家族主动脉疾病的病因。对八名受影响的成年人进行评估,未发现马凡综合征的主要骨骼表现。家族hMFS002在外显子37有一个错义突变(R1530C)。四名成员符合眼部和心血管表型标准,但缺乏骨骼表现。家族hMFS003在外显子12有两个连续的错义FBN1突变(C515W和R516G)。八名成员符合马凡综合征的眼部标准,两名成员有主要心血管表现,但他们均不符合骨骼系统标准。这些数据表明,具有FBN1突变的西班牙裔个体可能不会像白种人那样表现出马凡综合征的骨骼特征。我们建议,即使没有骨骼特征,包括西班牙裔患者在内的任何可能患有马凡综合征的患者都应考虑进行超声心动图、眼部检查和FBN1分子检测。

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