Literature DB >> 9263339

Ascending aortic aneurysm with or without features of Marfan syndrome and other fibrillinopathies: new insights.

H Furthmayr1, U Francke.   

Abstract

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.

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Year:  1997        PMID: 9263339

Source DB:  PubMed          Journal:  Semin Thorac Cardiovasc Surg        ISSN: 1043-0679


  4 in total

1.  Toward an understanding of the cause of mitral valve prolapse.

Authors:  J A Towbin
Journal:  Am J Hum Genet       Date:  1999-11       Impact factor: 11.025

2.  Cysteine substitutions in epidermal growth factor-like domains of fibrillin-1: distinct effects on biochemical and clinical phenotypes.

Authors:  I Schrijver; W Liu; T Brenn; H Furthmayr; U Francke
Journal:  Am J Hum Genet       Date:  1999-10       Impact factor: 11.025

3.  Premature termination mutations in FBN1: distinct effects on differential allelic expression and on protein and clinical phenotypes.

Authors:  Iris Schrijver; Wanguo Liu; Raanan Odom; Thomas Brenn; Peter Oefner; Heinz Furthmayr; Uta Francke
Journal:  Am J Hum Genet       Date:  2002-06-14       Impact factor: 11.025

4.  Multi-exon deletions of the FBN1 gene in Marfan syndrome.

Authors:  W Liu; I Schrijver; T Brenn; H Furthmayr; U Francke
Journal:  BMC Med Genet       Date:  2001-10-24       Impact factor: 2.103

  4 in total

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