Literature DB >> 7351497

Dermatosparaxis in a Himalayan cat: II. Ultrastructural studies of dermal collagen.

K A Holbrook, P H Byers, D F Counts, G A Hegreberg.   

Abstract

Dermatosparaxis is a connective tissue disease, primarily of sheep and cattle, that results from deficient activity of the NH2-terminal procollagen peptidase. It is characterized by fragile, loose skin that is easily torn with minor trauma. We have identified a cat twith a defect in this procollagen peptidase which affects only a small proportion of the collagen molecules; the majority of the collagen is processed normally. Nonetheless, as seen by transmission and scanning electron microscopy, this population of aberrant collagen molecules significantly alters the structure of individual collagen fibrils, the assembly of fibrils into fiber bundles and the integration of fiber bundles into a normal, woven network in the reticular dermis of skin. Although the clinical findings are less severe than those in sheep and cattle where the enzymatic defect is more complete, the ultrastructural abnormalities are marked and demonstrate that a minority of abnormal collagen molecules cn have a major effect on the structure and function of connective tissues.

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Year:  1980        PMID: 7351497     DOI: 10.1111/1523-1747.ep12520000

Source DB:  PubMed          Journal:  J Invest Dermatol        ISSN: 0022-202X            Impact factor:   8.551


  13 in total

1.  Human dermatosparaxis: a form of Ehlers-Danlos syndrome that results from failure to remove the amino-terminal propeptide of type I procollagen.

Authors:  L T Smith; W Wertelecki; L M Milstone; E M Petty; M R Seashore; I M Braverman; T G Jenkins; P H Byers
Journal:  Am J Hum Genet       Date:  1992-08       Impact factor: 11.025

Review 2.  Learning how mutations in type I collagen genes cause connective tissue disease.

Authors:  K E Kadler
Journal:  Int J Exp Pathol       Date:  1993-08       Impact factor: 1.925

3.  Alterations of collagen in lichen amyloidosus.

Authors:  D Tsambaos; R Bolling
Journal:  Arch Dermatol Res       Date:  1980       Impact factor: 3.017

4.  Processing of types I and III procollagen in Ehlers-Danlos syndrome type VII.

Authors:  R Halila; B Steinmann; L Peltonen
Journal:  Am J Hum Genet       Date:  1986-08       Impact factor: 11.025

5.  Differential ultrastructural aberrations of collagen fibrils in Ehlers-Danlos syndrome types I-IV as a means of diagnostics and classification.

Authors:  I Hausser; I Anton-Lamprecht
Journal:  Hum Genet       Date:  1994-04       Impact factor: 4.132

6.  The clinical features of Ehlers-Danlos syndrome type VIIB resulting from a base substitution at the splice acceptor site of intron 5 of the COL1A2 gene.

Authors:  A J Carr; A A Chiodo; J M Hilton; C W Chow; A Hockey; W G Cole
Journal:  J Med Genet       Date:  1994-04       Impact factor: 6.318

7.  Dermatosparaxis in two Limousin calves.

Authors:  Catherine I Carty; Alison M Lee; Nathan A E Wienandt; Edward L Stevens; Derron A Alves; John A Browne; Jill Bryan; Eoin G Ryan; Joseph P Cassidy
Journal:  Ir Vet J       Date:  2016-10-18       Impact factor: 2.146

8.  Surgical and medical treatment of ocular disease in a dog with Ehlers-Danlos syndrome.

Authors:  Søren N Rasch
Journal:  Clin Case Rep       Date:  2017-04-24

9.  A homozygous ADAMTS2 nonsense mutation in a Doberman Pinscher dog with Ehlers Danlos syndrome and extreme skin fragility.

Authors:  J A Jaffey; G Bullock; E Teplin; J Guo; N A Villani; T Mhlanga-Mutangadura; R D Schnabel; L A Cohn; G S Johnson
Journal:  Anim Genet       Date:  2019-07-11       Impact factor: 3.169

10.  Expanding the clinical and mutational spectrum of the Ehlers-Danlos syndrome, dermatosparaxis type.

Authors:  Tim Van Damme; Alain Colige; Delfien Syx; Cecilia Giunta; Uschi Lindert; Marianne Rohrbach; Omid Aryani; Yasemin Alanay; Pelin Özlem Simsek-Kiper; Hester Y Kroes; Koen Devriendt; Marc Thiry; Sofie Symoens; Anne De Paepe; Fransiska Malfait
Journal:  Genet Med       Date:  2016-01-14       Impact factor: 8.822

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