Literature DB >> 8541855

Linkage of the gene that encodes the alpha 1 chain of type V collagen (COL5A1) to type II Ehlers-Danlos syndrome (EDS II).

J Loughlin1, C Irven, L J Hardwick, S Butcher, S Walsh, P Wordsworth, B Sykes.   

Abstract

Ehlers-Danlos syndrome (EDS) is a group of heritable disorders of connective tissue with skin, ligaments and blood vessels being the main sites affected. The commonest variant (EDS II) exhibits an autosomal dominant mode of inheritance and is characterized by joint hypermobility, cigarette paper scars, lax skin and excessive bruising. As yet no gene has been linked to EDS II, nor has linkage been established to a specific region of the genome. However, several candidate genes encoding proteins of the extracellular matrix have been excluded. Using an intragenic simple sequence repeat polymorphism, we report linkage of the COL5A1 gene, which encodes the alpha 1(V) chain of type V collagen, to EDS II. A maximum LOD score (Zmax) for linkage of 8.3 at theta = 0.00 was generated for a single large pedigree.

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Year:  1995        PMID: 8541855     DOI: 10.1093/hmg/4.9.1649

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  8 in total

1.  Null alleles of the COL5A1 gene of type V collagen are a cause of the classical forms of Ehlers-Danlos syndrome (types I and II).

Authors:  U Schwarze; M Atkinson; G G Hoffman; D S Greenspan; P H Byers
Journal:  Am J Hum Genet       Date:  2000-05-04       Impact factor: 11.025

2.  Periostin regulates collagen fibrillogenesis and the biomechanical properties of connective tissues.

Authors:  Russell A Norris; Brook Damon; Vladimir Mironov; Vladimir Kasyanov; Anand Ramamurthi; Ricardo Moreno-Rodriguez; Thomas Trusk; Jay D Potts; Richard L Goodwin; Jeff Davis; Stanley Hoffman; Xuejun Wen; Yukiko Sugi; Christine B Kern; Corey H Mjaatvedt; Debi K Turner; Toru Oka; Simon J Conway; Jeffery D Molkentin; Gabor Forgacs; Roger R Markwald
Journal:  J Cell Biochem       Date:  2007-06-01       Impact factor: 4.429

3.  Mutations in the COL5A1 gene are causal in the Ehlers-Danlos syndromes I and II.

Authors:  A De Paepe; L Nuytinck; I Hausser; I Anton-Lamprecht; J M Naeyaert
Journal:  Am J Hum Genet       Date:  1997-03       Impact factor: 11.025

Review 4.  Ehlers-Danlos syndrome has varied molecular mechanisms.

Authors:  F M Pope; N P Burrows
Journal:  J Med Genet       Date:  1997-05       Impact factor: 6.318

5.  An exon skipping mutation of a type V collagen gene (COL5A1) in Ehlers-Danlos syndrome.

Authors:  A C Nicholls; J E Oliver; S McCarron; J B Harrison; D S Greenspan; F M Pope
Journal:  J Med Genet       Date:  1996-11       Impact factor: 6.318

6.  Targeted disruption of decorin leads to abnormal collagen fibril morphology and skin fragility.

Authors:  K G Danielson; H Baribault; D F Holmes; H Graham; K E Kadler; R V Iozzo
Journal:  J Cell Biol       Date:  1997-02-10       Impact factor: 10.539

7.  A point mutation in an intronic branch site results in aberrant splicing of COL5A1 and in Ehlers-Danlos syndrome type II in two British families.

Authors:  N P Burrows; A C Nicholls; A J Richards; C Luccarini; J B Harrison; J R Yates; F M Pope
Journal:  Am J Hum Genet       Date:  1998-08       Impact factor: 11.025

8.  Identification of a de novo case of COL5A1-related Ehlers-Danlos syndrome in an infant in the West Indies leading to improved targeted clinical care.

Authors:  Amr Wardeh; Tyson Jackson; Beverly Nelson; Carl Ernst; Jean-François Théroux; Walla Al-Hertani; Andrew K Sobering; Mary C Maj
Journal:  Clin Case Rep       Date:  2018-10-15
  8 in total

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