Literature DB >> 429005

Biochemical characteristics of Ehlers-Danlos syndrome type VI in a family with one affected infant.

T Krieg, U Feldmann, W Kessler, P K Müller.   

Abstract

The parents of a child with the clinical symptoms of Ehlers-Danlos syndrome type VI were identified as third-degree cousins. Biochemical analysis of the dermis of the patient revealed a complete lack of hydroxylysine in the dermal collagen. The dermis of both parents contained only half the amount of hydroxylysine found in healthy individuals. Hydroxylation of prolyl residues was normal in the skin of the patient and his parents. Investigation of the collagen synthesized by fibroblasts derived from the skin of the patient showed a normal proportion of type I and type III collagen. However, while hydroxylation of prolyl residues was normal in type I and type III collagen, hydroxylation of lysyl residues was markedly lower than normal in both type I and type III collagen.

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Year:  1979        PMID: 429005     DOI: 10.1007/bf00278900

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  12 in total

1.  Ehlers-Danlos syndrome in two siblings with deficient lysyl hydroxylase activity in cultured skin fibroblasts but only mild hydroxylysine deficit in skin.

Authors:  B Steinmann; R Gitzelmann; A Vogel; M E Grant; R Harwood; C H Sear
Journal:  Helv Paediatr Acta       Date:  1975-10

2.  Biochemical characteristics and biological significance of the genetically-distinct collagens.

Authors:  E J Miller
Journal:  Mol Cell Biochem       Date:  1976-12-10       Impact factor: 3.396

3.  Ascorbate-dependent differences in the hydroxylation of proline and lysine in collagen synthesized by 3T6 fibroblasts in culture.

Authors:  C J Bates; C J Prynne; C I Levene
Journal:  Biochim Biophys Acta       Date:  1972-10-31

4.  A study on the conversion of procollagen. Release and recovery of procollagen peptides in the culture medium.

Authors:  B F Pontz; P K Müller; W N Meigel
Journal:  J Biol Chem       Date:  1973-11-10       Impact factor: 5.157

5.  A heritable disorder of connective tissue. Hydroxylysine-deficient collagen disease.

Authors:  S R Pinnell; S M Krane; J E Kenzora; M J Glimcher
Journal:  N Engl J Med       Date:  1972-05-11       Impact factor: 91.245

6.  Abnormal properties of collagen lysyl hydroxylase from skin fibroblasts of siblings with hydroxylysine-deficient collagen.

Authors:  R S Quinn; S M Krane
Journal:  J Clin Invest       Date:  1976-01       Impact factor: 14.808

7.  Lysyl-protocollagen hydroxylase deficiency in fibroblasts from siblings with hydroxylysine-deficient collagen.

Authors:  S M Krane; S R Pinnell; R W Erbe
Journal:  Proc Natl Acad Sci U S A       Date:  1972-10       Impact factor: 11.205

8.  Hydroxylysine-deficient skin collagen in a patient with a form of the Ehlers-Danlos syndrome.

Authors:  M Sussman; J R Lichtenstein; T P Nigra; G R Martin; V A McKusick
Journal:  J Bone Joint Surg Am       Date:  1974-09       Impact factor: 5.284

9.  The nature of the collagen synthesized by cultured human fibroblasts.

Authors:  D L Layman; E B McGoodwin; G R Martin
Journal:  Proc Natl Acad Sci U S A       Date:  1971-02       Impact factor: 11.205

10.  Inherited human collagen lysyl hydroxylase deficiency: ascorbic acid response.

Authors:  L J Elsas; R L Miller; S R Pinnell
Journal:  J Pediatr       Date:  1978-03       Impact factor: 4.406

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  6 in total

1.  Lysyl hydroxylation in collagens from hyperplastic callus and embryonic bones.

Authors:  H W Lehmann; M Bodo; C Frohn; A Nerlich; D Rimek; H Notbohm; P K Müller
Journal:  Biochem J       Date:  1992-03-01       Impact factor: 3.857

2.  Genotyping and prenatal assessment of collagen lysyl hydroxylase deficiency in a family with Ehlers-Danlos syndrome type VI.

Authors:  P P Dembure; J H Priest; S C Snoddy; L J Elsas
Journal:  Am J Hum Genet       Date:  1984-07       Impact factor: 11.025

3.  Osteogenesis imperfecta: biochemical and clinical evaluation of 13 cases.

Authors:  T Krieg; E Kirsch; K Matzen; P K Müller
Journal:  Klin Wochenschr       Date:  1981-01-15

4.  Collagen biosynthesis and isomorphism in a case of Ehlers-Danlos syndrome type VI.

Authors:  A Chamson; P Berbis; J F Fabre; Y Privat; J Frey
Journal:  Arch Dermatol Res       Date:  1987       Impact factor: 3.017

5.  Alu-Alu recombination results in a duplication of seven exons in the lysyl hydroxylase gene in a patient with the type VI variant of Ehlers-Danlos syndrome.

Authors:  B Pousi; T Hautala; J Heikkinen; L Pajunen; K I Kivirikko; R Myllylä
Journal:  Am J Hum Genet       Date:  1994-11       Impact factor: 11.025

6.  Skin malformations in a neonatal foal tested homozygous positive for Warmblood Fragile Foal Syndrome.

Authors:  Chloé Monthoux; Simone de Brot; Michelle Jackson; Ulrich Bleul; Jasmin Walter
Journal:  BMC Vet Res       Date:  2015-01-31       Impact factor: 2.741

  6 in total

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