Literature DB >> 7691719

Fibrillin binds calcium and is coded by cDNAs that reveal a multidomain structure and alternatively spliced exons at the 5' end.

G M Corson1, S C Chalberg, H C Dietz, N L Charbonneau, L Y Sakai.   

Abstract

Fibrillin is an important structural protein of the extracellular matrix. It is a large cysteine-rich glycoprotein with extensive intrachain disulfide bonds, likely contributed by multiple EGF-like repeats. We have previously published 6.9 kb of FBN1 cDNA sequence. FBN1 cDNA clones that extend the sequence 3089 bp in the 5' direction are described in this report. The deduced primary structure suggests that fibrillin is composed of multiple domains. The most predominant feature is the presence of 43 calcium binding EGF-like repeats. We demonstrate here that fibrillin molecules bind calcium. In addition, three alternatively spliced exons at the 5' end are described. Analysis of 5.8 kb of surrounding genomic sequence revealed a 1.8-kb CpG island spanning the alternatively spliced exons and the next downstream exon. Since FBN1 is the gene responsible for Marfan syndrome, the information presented here will be useful in identifying new mutations and in understanding the function of fibrillin in the pathogenesis of the disease.

Entities:  

Mesh:

Substances:

Year:  1993        PMID: 7691719     DOI: 10.1006/geno.1993.1350

Source DB:  PubMed          Journal:  Genomics        ISSN: 0888-7543            Impact factor:   5.736


  66 in total

1.  Fibrillin degradation by matrix metalloproteinases: implications for connective tissue remodelling.

Authors:  J L Ashworth; G Murphy; M J Rock; M J Sherratt; S D Shapiro; C A Shuttleworth; C M Kielty
Journal:  Biochem J       Date:  1999-05-15       Impact factor: 3.857

2.  N-terminal domains of fibrillin 1 and fibrillin 2 direct the formation of homodimers: a possible first step in microfibril assembly.

Authors:  T M Trask; T M Ritty; T Broekelmann; C Tisdale; R P Mecham
Journal:  Biochem J       Date:  1999-06-15       Impact factor: 3.857

3.  Fibrillin-containing microfibrils are key signal relay stations for cell function.

Authors:  Karina A Zeyer; Dieter P Reinhardt
Journal:  J Cell Commun Signal       Date:  2015-10-08       Impact factor: 5.782

Review 4.  The molecular genetics of Marfan syndrome and related microfibrillopathies.

Authors:  P N Robinson; M Godfrey
Journal:  J Med Genet       Date:  2000-01       Impact factor: 6.318

Review 5.  Marfan's syndrome.

Authors:  Daniel P Judge; Harry C Dietz
Journal:  Lancet       Date:  2005-12-03       Impact factor: 79.321

6.  Marfan Database (second edition): software and database for the analysis of mutations in the human FBN1 gene.

Authors:  G Collod-Béroud; C Béroud; L Adès; C Black; M Boxer; D J Brock; M Godfrey; C Hayward; L Karttunen; D Milewicz; L Peltonen; R I Richards; M Wang; C Junien; C Boileau
Journal:  Nucleic Acids Res       Date:  1997-01-01       Impact factor: 16.971

7.  Of mice and Marfan: genetic linkage analyses of the fibrillin genes, Fbn1 and Fbn2, in the mouse genome.

Authors:  C Goldstein; P Liaw; S A Jimenez; A M Buchberg; L D Siracusa
Journal:  Mamm Genome       Date:  1994-11       Impact factor: 2.957

8.  Early fibrillin-1 assembly monitored through a modifiable recombinant cell approach.

Authors:  Dirk Hubmacher; Eric Bergeron; Christine Fagotto-Kaufmann; Lynn Y Sakai; Dieter P Reinhardt
Journal:  Biomacromolecules       Date:  2014-03-07       Impact factor: 6.988

Review 9.  Identification of defects in the fibrillin gene and protein in individuals with the Marfan syndrome and related disorders.

Authors:  D M Milewicz
Journal:  Tex Heart Inst J       Date:  1994

10.  Identification of the minimal combination of clinical features in probands for efficient mutation detection in the FBN1 gene.

Authors:  Chantal Stheneur; Gwenaëlle Collod-Béroud; Laurence Faivre; Jean François Buyck; Laurent Gouya; Jean-Marie Le Parc; Bertrand Moura; Christine Muti; Bernard Grandchamp; Gilles Sultan; Mireille Claustres; Philippe Aegerter; Bertrand Chevallier; Guillaume Jondeau; Catherine Boileau
Journal:  Eur J Hum Genet       Date:  2009-03-18       Impact factor: 4.246

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.