Literature DB >> 12723256

Genetic fibrillinopathies: new insights in molecular diagnosis and clinical management.

B L Loeys, D M Matthys, A M de Paepe.   

Abstract

The Marfan syndrome (MFS) is an autosomal dominant connective tissue disorder with a prevalence of 2-3 per 10,000 individuals and symptoms ranging from skeletal overgrowth, cutaneous striae to ectopia lentis and aortic dilatation leading to dissection. Mutation in the gene for fibrillin-1 (FBN1) cause MFS and other related disorders of connective tissue, grouped as fibrillinopathies. Fibrillin-1 is the main constituent of extracellular microfibrils. Microfibrils can exist as individual structures or associate with elastin to form elastic fibers. This article provides an overview of the current diagnostic criteria and medical management, estimates the role of fibrillin-1 mutation analysis, sheds new light on genotype-phenotype correlations and summarizes new insights on the pathogenesis of this disorder based on mouse models.

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Year:  2003        PMID: 12723256     DOI: 10.1179/acb.2003.58.1.001

Source DB:  PubMed          Journal:  Acta Clin Belg        ISSN: 1784-3286            Impact factor:   1.264


  5 in total

1.  Spontaneous pneumothorax.

Authors:  Heinrich Matthys
Journal:  Multidiscip Respir Med       Date:  2011-02-28

2.  Functional genomic analysis of C. elegans molting.

Authors:  Alison R Frand; Sascha Russel; Gary Ruvkun
Journal:  PLoS Biol       Date:  2005-08-30       Impact factor: 8.029

3.  Prevalence of scoliosis in Williams-Beuren syndrome patients treated at a regional reference center.

Authors:  Marcelo Loquette Damasceno; Alexandre Fogaça Cristante; Raphael Martus Marcon; Tarcísio Eloy Pessoa de Barros Filho
Journal:  Clinics (Sao Paulo)       Date:  2014-07       Impact factor: 2.365

4.  Extracellular Regulation of Bone Morphogenetic Protein Activity by the Microfibril Component Fibrillin-1.

Authors:  Alexander P Wohl; Helen Troilo; Richard F Collins; Clair Baldock; Gerhard Sengle
Journal:  J Biol Chem       Date:  2016-04-08       Impact factor: 5.157

5.  Case Report: A Novel Homozygous Missense Variant of FBN3 Supporting It Is a New Candidate Gene Causative of a Bardet-Biedl Syndrome-Like Phenotype.

Authors:  Maria Luce Genovesi; Barbara Torres; Marina Goldoni; Eliana Salvo; Claudia Cesario; Massimo Majolo; Tommaso Mazza; Carmelo Piscopo; Laura Bernardini
Journal:  Front Genet       Date:  2022-07-15       Impact factor: 4.772

  5 in total

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