Literature DB >> 8411057

The substitution of glycine 661 by arginine in type III collagen produces mutant molecules with different thermal stabilities and causes Ehlers-Danlos syndrome type IV.

A Richards1, P Narcisi, J Lloyd, C Ferguson, F M Pope.   

Abstract

Previous studies have shown that Ehlers-Danlos syndrome type IV (EDS IV) is caused by mutations of type III collagen (COL3A1). Here we have characterised the most amino-terminal glycine substitution so far described in a patient with EDS IV. A combination of peptide mapping and chemical cleavage analysis of cDNA localised the mutation in cyanogen bromide peptide CB5. Sequence analysis showed a G to A mutation, converting glycine 661 to arginine, which was a new dominant mutation. Analysis of type III collagen secreted by cultured fibroblasts showed an overmodified mutant protein with normal thermal stability. However, the intracellularly retained form melted 2 degrees C lower than normal. This indicated that molecules resulting from the same mutation can differ in their thermal stabilities.

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Year:  1993        PMID: 8411057      PMCID: PMC1016501          DOI: 10.1136/jmg.30.8.690

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  13 in total

1.  A 27-bp deletion from one allele of the type III collagen gene (COL3A1) in a large family with Ehlers-Danlos syndrome type IV.

Authors:  A J Richards; J C Lloyd; P Narcisi; P N Ward; A C Nicholls; A De Paepe; F M Pope
Journal:  Hum Genet       Date:  1992-01       Impact factor: 4.132

2.  Linkage of a polymorphic marker for the type III collagen gene (COL3A1) to atypical autosomal dominant Ehlers-Danlos syndrome type IV in a large Belgian pedigree.

Authors:  A C Nicholls; A De Paepe; P Narcisi; R Dalgleish; F De Keyser; M Matton; F M Pope
Journal:  Hum Genet       Date:  1988-03       Impact factor: 4.132

3.  Synthesis of an altered type III procollagen in a patient with type IV Ehlers-Danlos syndrome. A structural change in the alpha 1(III) chain which makes the protein more susceptible to proteinases.

Authors:  C A Stolle; R E Pyeritz; J C Myers; D J Prockop
Journal:  J Biol Chem       Date:  1985-02-10       Impact factor: 5.157

4.  Single base mutation in the type III procollagen gene that converts the codon for glycine 883 to aspartate in a mild variant of Ehlers-Danlos syndrome IV.

Authors:  G Tromp; H Kuivaniemi; C Stolle; F M Pope; D J Prockop
Journal:  J Biol Chem       Date:  1989-11-15       Impact factor: 5.157

5.  A single base mutation in the gene for type III collagen (COL3A1) converts glycine 847 to glutamic acid in a family with Ehlers-Danlos syndrome type IV. An unaffected family member is mosaic for the mutation.

Authors:  A J Richards; P N Ward; P Narcisi; A C Nicholls; J C Lloyd; F M Pope
Journal:  Hum Genet       Date:  1992-06       Impact factor: 4.132

6.  Single base mutation that substitutes glutamic acid for glycine 1021 in the COL3A1 gene and causes Ehlers-Danlos syndrome type IV.

Authors:  P Narcisi; Y Wu; G Tromp; J J Earley; A J Richards; F M Pope; H Kuivaniemi
Journal:  Am J Med Genet       Date:  1993-05-15

7.  Characterisation of a glycine to valine substitution at amino acid position 910 of the triple helical region of type III collagen in a patient with Ehlers-Danlos syndrome type IV.

Authors:  A J Richards; J C Lloyd; P N Ward; A De Paepe; P Narcisi; F M Pope
Journal:  J Med Genet       Date:  1991-07       Impact factor: 6.318

8.  Structure of cDNA clones coding for the entire prepro alpha 1 (III) chain of human type III procollagen. Differences in protein structure from type I procollagen and conservation of codon preferences.

Authors:  L Ala-Kokko; S Kontusaari; C T Baldwin; H Kuivaniemi; D J Prockop
Journal:  Biochem J       Date:  1989-06-01       Impact factor: 3.857

9.  Subtle structural alterations in the chains of type I procollagen produce osteogenesis imperfecta type II.

Authors:  J Bonadio; P H Byers
Journal:  Nature       Date:  1985 Jul 25-31       Impact factor: 49.962

10.  Patients with Ehlers-Danlos syndrome type IV lack type III collagen.

Authors:  F M Pope; G R Martin; J R Lichtenstein; R Penttinen; B Gerson; D W Rowe; V A McKusick
Journal:  Proc Natl Acad Sci U S A       Date:  1975-04       Impact factor: 11.205

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  4 in total

1.  The G2385R variant of leucine-rich repeat kinase 2 associated with Parkinson's disease is a partial loss-of-function mutation.

Authors:  Iakov N Rudenko; Alice Kaganovich; David N Hauser; Aleksandra Beylina; Ruth Chia; Jinhui Ding; Dragan Maric; Howard Jaffe; Mark R Cookson
Journal:  Biochem J       Date:  2012-08-15       Impact factor: 3.857

Review 2.  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

Review 3.  Molecular Basis of Pathogenic Variants in the Fibrillar Collagens.

Authors:  Allan J Richards; Martin P Snead
Journal:  Genes (Basel)       Date:  2022-07-04       Impact factor: 4.141

Review 4.  Ehlers-Danlos syndrome type IV.

Authors:  Dominique P Germain
Journal:  Orphanet J Rare Dis       Date:  2007-07-19       Impact factor: 4.123

  4 in total

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