Literature DB >> 11706995

The molecular pathogenesis of the Marfan syndrome.

P N Robinson1, P Booms.   

Abstract

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.

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Year:  2001        PMID: 11706995     DOI: 10.1007/pl00000807

Source DB:  PubMed          Journal:  Cell Mol Life Sci        ISSN: 1420-682X            Impact factor:   9.261


  13 in total

1.  Functional role of periostin in development and wound repair: implications for connective tissue disease.

Authors:  Douglas W Hamilton
Journal:  J Cell Commun Signal       Date:  2008-07-20       Impact factor: 5.782

Review 2.  Extracellular matrix molecules: potential targets in pharmacotherapy.

Authors:  Hannu Järveläinen; Annele Sainio; Markku Koulu; Thomas N Wight; Risto Penttinen
Journal:  Pharmacol Rev       Date:  2009-06       Impact factor: 25.468

Review 3.  Tissue elasticity and the ageing elastic fibre.

Authors:  Michael J Sherratt
Journal:  Age (Dordr)       Date:  2009-12

Review 4.  The dynamic sclera: extracellular matrix remodeling in normal ocular growth and myopia development.

Authors:  Angelica R Harper; Jody A Summers
Journal:  Exp Eye Res       Date:  2015-04       Impact factor: 3.467

5.  Copper-beaten skull appearance in the setting of Marfan syndrome.

Authors:  Vladimir Živković; Danica Cvetković; Slobodan Nikolić
Journal:  Forensic Sci Med Pathol       Date:  2017-10-23       Impact factor: 2.007

Review 6.  The molecular genetics of Marfan syndrome and related disorders.

Authors:  P N Robinson; E Arteaga-Solis; C Baldock; G Collod-Béroud; P Booms; A De Paepe; H C Dietz; G Guo; P A Handford; D P Judge; C M Kielty; B Loeys; D M Milewicz; A Ney; F Ramirez; D P Reinhardt; K Tiedemann; P Whiteman; M Godfrey
Journal:  J Med Genet       Date:  2006-03-29       Impact factor: 6.318

7.  Characterization of the renal phenotype in a mouse model of Marfan syndrome.

Authors:  Andrea Hartner; Timo Eifert; Christian S Haas; Cigdem Tuysuz; Karl F Hilgers; Dieter P Reinhardt; Kerstin Amann
Journal:  Virchows Arch       Date:  2004-07-29       Impact factor: 4.064

8.  Enumeration of the colony-forming units-fibroblast from mouse and human bone marrow in normal and pathological conditions.

Authors:  Sergei A Kuznetsov; Mahesh H Mankani; Paolo Bianco; Pamela G Robey
Journal:  Stem Cell Res       Date:  2008-08-12       Impact factor: 2.020

9.  Marfan syndrome: clinical consequences resulting from a medicolegal autopsy of a case of sudden death due to aortic rupture.

Authors:  M Klintschar; U Bilkenroth; M Arslan-Kirchner; J Schmidtke; D Stiller
Journal:  Int J Legal Med       Date:  2008-10-17       Impact factor: 2.686

10.  Induction of macrophage chemotaxis by aortic extracts from patients with Marfan syndrome is related to elastin binding protein.

Authors:  Gao Guo; Petra Gehle; Sandra Doelken; José Luis Martin-Ventura; Yskert von Kodolitsch; Roland Hetzer; Peter N Robinson
Journal:  PLoS One       Date:  2011-05-27       Impact factor: 3.240

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