Mutations of the gene encoding the transmembrane transporter protein ABC-C6 cause pseudoxanthoma elasticum

Details

Serval ID
serval:BIB_85B1952165E6
Type
Article: article from journal or magazin.
Collection
Publications
Institution
Title
Mutations of the gene encoding the transmembrane transporter protein ABC-C6 cause pseudoxanthoma elasticum
Journal
Journal of Molecular Medicine
Author(s)
Struk  B., Cai  L., Zach  S., Ji  W., Chung  J., Lumsden  A., Stumm  M., Huber  M., Schaen  L., Kim  C. A., Goldsmith  L. A., Viljoen  D., Figuera  L. E., Fuchs  W., Munier  F., Ramesar  R., Hohl  D., Richards  R., Neldner  K. H., Lindpaintner  K.
ISSN
0946-2716 (Print)
Publication state
Published
Issued date
2000
Volume
78
Number
5
Pages
282-6
Notes
Journal Article
Research Support, Non-U.S. Gov't
Research Support, U.S. Gov't, P.H.S.
Abstract
We recently published the precise chromosomal localization on chromosome 16p13.1 of the genetic defect underlying pseudoxanthoma elasticum (PXE), an inherited disorder characterized by progressive calcification of elastic fibers in skin, eye, and the cardiovascular system. Here we report the identification of mutations in the gene encoding the transmembrane transporter protein, ABC-C6 (also known as MRP-6), one of the four genes located in the region of linkage, as cause of the disease. Sequence analysis in four independent consanguineous families from Switzerland, Mexico, and South Africa and in one non-consanguineous family from the United States demonstrated several different mis-sense mutations to cosegregate with the disease phenotype. These findings are consistent with the conclusion that PXE is a recessive disorder that displays allelic heterogeneity, which may explain the considerable phenotypic variance characteristic of the disorder.
Keywords
ATP-Binding Cassette Transporters/*genetics Consanguinity Female Haplotypes/genetics Homozygote Humans Male Multidrug Resistance-Associated Proteins *Mutation Pedigree Point Mutation Polymorphism, Restriction Fragment Length Pseudoxanthoma Elasticum/*genetics
Pubmed
Web of science
Create date
28/01/2008 13:54
Last modification date
20/08/2019 15:45
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