Literature DB >> 15859521

Defective glycosaminoglycan substitution of decorin in a patient with progeroid syndrome is a direct consequence of two point mutations in the galactosyltransferase I (beta4GalT-7) gene.

Martin Götte1, Hans Kresse.   

Abstract

The small dermatan sulfate proteoglycan decorin is involved in the regulation of collagen fibrillogenesis, cell adhesion and migration, and growth factor signaling. In a progeroid patient carrying two point mutations in beta4 galactosyltransferase I (beta4GalT-7) only 50% of the decorin core protein molecules are substituted with glycosaminoglycan chains. We expressed decorin, as well as wild-type and mutant alleles of beta4GalT-7 in galactosyltransferase-deficient CHO618 cells. Decorin was less efficiently substituted with glycosaminoglycan chains upon expression of beta4GalT-7(186D) compared to beta4GalT-7-expressing cells. Decorin from beta4GalT-7-expressing cells displayed increased molecular heterogeneity. Decorin glycosaminoglycan chains were completely susceptible to chondroitinase ABC treatment. Cells expressing beta4GalT-7(206P) did not synthesize the proteoglycanform of decorin. Thus, the beta4GalT-7 mutations directly affect the molecular phenotype of decorin observed in a patient with the progeroid form of Ehlers-Danlos syndrome, which may be a major mechanistic cause for the skin and wound healing defects observed in this patient.

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Year:  2005        PMID: 15859521     DOI: 10.1007/s10528-005-1068-2

Source DB:  PubMed          Journal:  Biochem Genet        ISSN: 0006-2928            Impact factor:   1.890


  18 in total

Review 1.  Golgi glycosylation and human inherited diseases.

Authors:  Hudson H Freeze; Bobby G Ng
Journal:  Cold Spring Harb Perspect Biol       Date:  2011-09-01       Impact factor: 10.005

Review 2.  Redefining the progeroid form of Ehlers-Danlos syndrome: report of the fourth patient with B4GALT7 deficiency and review of the literature.

Authors:  Michael H Guo; Joan Stoler; Julian Lui; Ola Nilsson; Diana W Bianchi; Joel N Hirschhorn; Andrew Dauber
Journal:  Am J Med Genet A       Date:  2013-08-16       Impact factor: 2.802

Review 3.  Heparan sulfate proteoglycans.

Authors:  Stephane Sarrazin; William C Lamanna; Jeffrey D Esko
Journal:  Cold Spring Harb Perspect Biol       Date:  2011-07-01       Impact factor: 10.005

4.  Lumican deficiency results in cardiomyocyte hypertrophy with altered collagen assembly.

Authors:  Loren E Dupuis; Matthew G Berger; Samuel Feldman; Lorna Doucette; Vennece Fowlkes; Shukti Chakravarti; Sarah Thibaudeau; Nicolas E Alcala; Amy D Bradshaw; Christine B Kern
Journal:  J Mol Cell Cardiol       Date:  2015-04-15       Impact factor: 5.000

5.  Chondrodysplasia and abnormal joint development associated with mutations in IMPAD1, encoding the Golgi-resident nucleotide phosphatase, gPAPP.

Authors:  Lisenka E L M Vissers; Ekkehart Lausch; Sheila Unger; Ana Belinda Campos-Xavier; Christian Gilissen; Antonio Rossi; Marisol Del Rosario; Hanka Venselaar; Ute Knoll; Sheela Nampoothiri; Mohandas Nair; Jürgen Spranger; Han G Brunner; Luisa Bonafé; Joris A Veltman; Bernhard Zabel; Andrea Superti-Furga
Journal:  Am J Hum Genet       Date:  2011-05-05       Impact factor: 11.025

6.  Defective glycosylation of decorin and biglycan, altered collagen structure, and abnormal phenotype of the skin fibroblasts of an Ehlers-Danlos syndrome patient carrying the novel Arg270Cys substitution in galactosyltransferase I (beta4GalT-7).

Authors:  Daniela G Seidler; Muhammad Faiyaz-Ul-Haque; Uwe Hansen; George W Yip; Syed H E Zaidi; Ahmad S Teebi; Ludwig Kiesel; Martin Götte
Journal:  J Mol Med (Berl)       Date:  2006-04-01       Impact factor: 4.599

Review 7.  Human genetic disorders caused by mutations in genes encoding biosynthetic enzymes for sulfated glycosaminoglycans.

Authors:  Shuji Mizumoto; Shiro Ikegawa; Kazuyuki Sugahara
Journal:  J Biol Chem       Date:  2013-03-01       Impact factor: 5.157

Review 8.  The regulatory roles of small leucine-rich proteoglycans in extracellular matrix assembly.

Authors:  Shoujun Chen; David E Birk
Journal:  FEBS J       Date:  2013-02-14       Impact factor: 5.542

9.  Age-dependent alterations of decorin glycosaminoglycans in human skin.

Authors:  Yong Li; Ying Liu; Wei Xia; Dan Lei; John J Voorhees; Gary J Fisher
Journal:  Sci Rep       Date:  2013       Impact factor: 4.379

Review 10.  Mutations in Biosynthetic Enzymes for the Protein Linker Region of Chondroitin/Dermatan/Heparan Sulfate Cause Skeletal and Skin Dysplasias.

Authors:  Shuji Mizumoto; Shuhei Yamada; Kazuyuki Sugahara
Journal:  Biomed Res Int       Date:  2015-10-25       Impact factor: 3.411

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