| Literature DB >> 8411057 |
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.Entities:
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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