Literature DB >> 4125715

Synthesis and degradation of hyaluronic acid in the cultured fibroblasts of Marfan's disease.

S I Lamberg, A Dorfman.   

Abstract

Increased amounts of hyaluronic acid accumulate in fibroblasts cultured from patients with Marfan's disease, an autosomal dominant disorder. In the recessive Hurler's disease, the storage of glycosaminoglycan (GAG) is due to impaired degradation. This study examines the kinetics of GAG accumulation in Marfan's disease in order to determine whether the mechanism of accumulation differs from that in Hurler's disease.Marfan-derived fibroblasts incorporated [(14)C]acetate or [(14)C]glucosamine into GAG to a level 4-6 times greater than control fibroblasts. Sugar analyses, electrophoretic mobility, and enzyme susceptibility studies showed that the isolated material was hyaluronic acid. There were no differences in activity of a variety of glycosidases between Marfan and control fibroblasts, nor were there differences in the ability to degrade prelabeled hyaluronate by cell-free extracts. Finally, chase experiments showed parallel rates of loss of labeled GAG from control fibroblasts and fibroblasts from Marfan patients. It appears that hyaluronic acid was accumulating in greater amounts in the fibroblasts from patients with Marfan's disease because of a greater rate of synthesis as opposed to a decreased rate of breakdown.

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Year:  1973        PMID: 4125715      PMCID: PMC302501          DOI: 10.1172/JCI107433

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  24 in total

1.  A study of the conditions and mechanism of the diphenylamine reaction for the colorimetric estimation of deoxyribonucleic acid.

Authors:  K BURTON
Journal:  Biochem J       Date:  1956-02       Impact factor: 3.857

2.  The accumulation of hyaluronic acid in cultured fibroblasts of the Marfan syndrome.

Authors:  R Matalon; A Dorfman
Journal:  Biochem Biophys Res Commun       Date:  1968-07-26       Impact factor: 3.575

3.  The defect in Hurler's and Hunter's syndromes: faulty degradation of mucopolysaccharide.

Authors:  J C Fratantoni; C W Hall; E F Neufeld
Journal:  Proc Natl Acad Sci U S A       Date:  1968-06       Impact factor: 11.205

4.  Hurler's syndrome: biosynthesis of acid mucopolysaccharides in tissue culture.

Authors:  R Matalon; A Dorfman
Journal:  Proc Natl Acad Sci U S A       Date:  1966-10       Impact factor: 11.205

5.  Purification and properties of bacterial chondroitinases and chondrosulfatases.

Authors:  T Yamagata; H Saito; O Habuchi; S Suzuki
Journal:  J Biol Chem       Date:  1968-04-10       Impact factor: 5.157

6.  Partial inhibition by cycloheximide of haluronate synthesis in cell culture.

Authors:  C Smith; D Hamerman
Journal:  Proc Soc Exp Biol Med       Date:  1968-04

7.  Collagen metabolism of the skin in Marfan's syndrome.

Authors:  O Laitinen; J Uitto; M Iivanainen; M Hannuksela; K I Kivirikko
Journal:  Clin Chim Acta       Date:  1968-09       Impact factor: 3.786

8.  Tissue cultures and mycoplasmas.

Authors:  L Hayflick
Journal:  Tex Rep Biol Med       Date:  1965-06

9.  Study of fibroblasts in Marfan's syndrome.

Authors:  M Macek; J Hurych; M Chvapil; V Kadlecová
Journal:  Humangenetik       Date:  1966

10.  Hurler's syndrome. A genetic study in cell culture.

Authors:  B S Danes; A G Bearn
Journal:  J Exp Med       Date:  1966-01-01       Impact factor: 14.307

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  11 in total

1.  Marfan syndrome: defective synthesis, secretion, and extracellular matrix formation of fibrillin by cultured dermal fibroblasts.

Authors:  D M Milewicz; R E Pyeritz; E S Crawford; P H Byers
Journal:  J Clin Invest       Date:  1992-01       Impact factor: 14.808

2.  Evidence for rapid metabolic turnover of hyaluronate synthetase in Swarm rat chondrosarcoma chondrocytes.

Authors:  M K Bansal; R M Mason
Journal:  Biochem J       Date:  1986-06-01       Impact factor: 3.857

3.  Unilateral microfibrillar abnormalities in a case of asymmetric Marfan syndrome.

Authors:  M Godfrey; S Olson; R G Burgio; A Martini; M Valli; G Cetta; H Hori; D W Hollister
Journal:  Am J Hum Genet       Date:  1990-04       Impact factor: 11.025

4.  Cosegregation of elastin-associated microfibrillar abnormalities with the Marfan phenotype in families.

Authors:  M Godfrey; V Menashe; R G Weleber; R D Koler; R H Bigley; E Lovrien; J Zonana; D W Hollister
Journal:  Am J Hum Genet       Date:  1990-04       Impact factor: 11.025

5.  n-Butyrate inhibition of hyaluronate synthesis in cultured human fibroblasts.

Authors:  T J Smith
Journal:  J Clin Invest       Date:  1987-05       Impact factor: 14.808

6.  The Marfan syndrome--analysis of growth and cardiovascular manifestation.

Authors:  U Vetter; R Mayerhofer; D Lang; G von Bernuth; M B Ranke; A A Schmaltz
Journal:  Eur J Pediatr       Date:  1990-04       Impact factor: 3.183

7.  Marfan syndrome: abnormal alpha 2 chain in type I collagen.

Authors:  P H Byers; R C Siegel; K E Peterson; D W Rowe; K A Holbrook; L T Smith; Y H Chang; J C Fu
Journal:  Proc Natl Acad Sci U S A       Date:  1981-12       Impact factor: 11.205

8.  Regulation of glycosaminoglycan synthesis by thyroid hormone in vitro.

Authors:  T J Smith; Y Murata; A L Horwitz; L Philipson; S Refetoff
Journal:  J Clin Invest       Date:  1982-11       Impact factor: 14.808

9.  Homozygosity for autosomal dominant Marfan syndrome.

Authors:  J Chemke; R Nisani; A Feigl; R Garty; M Cooper; Y Bårash; D Duksin
Journal:  J Med Genet       Date:  1984-06       Impact factor: 6.318

10.  Dexamethasone regulation of glycosaminoglycan synthesis in cultured human skin fibroblasts. Similar effects of glucocorticoid and thyroid hormones.

Authors:  T J Smith
Journal:  J Clin Invest       Date:  1984-12       Impact factor: 14.808

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