Literature DB >> 3286309

Lack of type VII collagen in unaffected skin of patients with severe recessive dystrophic epidermolysis bullosa.

L Bruckner-Tuderman1, S Rüegger, B Odermatt, Y Mitsuhashi, U W Schnyder.   

Abstract

Type VII collagen, the major structural component of the anchoring fibrils, was assayed in normal unaffected skin of patients with different forms of hereditary epidermolysis bullosa. Immunofluorescence staining with affinity-purified polyclonal antibodies to type VII collagen revealed a complete absence of staining in the skin of patients with severe dystrophic recessive epidermolysis bullosa. In all other forms, localized recessive dystrophic, dominant dystrophic, junctional and simplex forms there was an intense continuous linear staining of type VII collagen at the dermoepidermal junction. Also, obligate heterozygote carriers of the gene for severe dystrophic recessive form showed a normal pattern of staining. As internal controls and to define the clinical diagnosis, staining with antibodies to type IV collagen, laminin and bullous pemphigoid antigen was also performed. All these antibodies showed a normal staining pattern indicating an intact general morphology of the dermoepidermal junction zone. These results suggest that there is a defect of type VII collagen in patients with severe recessive dystrophic epidermolysis bullosa. The data also suggest that the group of recessive dystrophic epidermolysis bullosa may be heterogeneous not only clinically, but also at the molecular level.

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Year:  1988        PMID: 3286309     DOI: 10.1159/000248673

Source DB:  PubMed          Journal:  Dermatologica        ISSN: 0011-9075


  11 in total

1.  Genetic linkage of recessive dystrophic epidermolysis bullosa to the type VII collagen gene.

Authors:  A Hovnanian; P Duquesnoy; C Blanchet-Bardon; R G Knowlton; S Amselem; M Lathrop; L Dubertret; J Uitto; M Goossens
Journal:  J Clin Invest       Date:  1992-09       Impact factor: 14.808

Review 2.  Keratin gene mutations in human skin disease.

Authors:  H P Stevens; M H Rustin
Journal:  Postgrad Med J       Date:  1994-11       Impact factor: 2.401

3.  Genetic linkage to the type VII collagen gene (COL7A1) in 26 families with generalised recessive dystrophic epidermolysis bullosa and anchoring fibril abnormalities.

Authors:  M G Dunnill; A J Richards; G Milana; F Mollica; D Atherton; I Winship; M Farrall; L al-Imara; R A Eady; F M Pope
Journal:  J Med Genet       Date:  1994-10       Impact factor: 6.318

Review 4.  Recessive dystrophic epidermolysis bullosa: a review of disease pathogenesis and update on future therapies.

Authors:  Luis Soro; Cynthia Bartus; Stephen Purcell
Journal:  J Clin Aesthet Dermatol       Date:  2015-05

5.  Identification of two splicing mutations in the collagen type VII gene (COL7A1) of a patient affected by the localisata variant of recessive dystrophic epidermolysis bullosa.

Authors:  R Gardella; L Belletti; N Zoppi; D Marini; S Barlati; M Colombi
Journal:  Am J Hum Genet       Date:  1996-08       Impact factor: 11.025

6.  Recurrent nonsense mutations within the type VII collagen gene in patients with severe recessive dystrophic epidermolysis bullosa.

Authors:  A Hovnanian; L Hilal; C Blanchet-Bardon; Y de Prost; A M Christiano; J Uitto; M Goossens
Journal:  Am J Hum Genet       Date:  1994-08       Impact factor: 11.025

Review 7.  The molecular basis for inherited bullous diseases.

Authors:  B P Korge; T Krieg
Journal:  J Mol Med (Berl)       Date:  1996-02       Impact factor: 4.599

8.  Exclusion of linkage between the collagenase gene and generalized recessive dystrophic epidermolysis bullosa phenotype.

Authors:  A Hovnanian; P Duquesnoy; S Amselem; C Blanchet-Bardon; M Lathrop; L Dubertret; M Goossens
Journal:  J Clin Invest       Date:  1991-11       Impact factor: 14.808

9.  Premature termination codons on both alleles of the type VII collagen gene (COL7A1) in three brothers with recessive dystrophic epidermolysis bullosa.

Authors:  A M Christiano; Y Suga; D S Greenspan; H Ogawa; J Uitto
Journal:  J Clin Invest       Date:  1995-03       Impact factor: 14.808

10.  180-kD bullous pemphigoid antigen (BP180) is deficient in generalized atrophic benign epidermolysis bullosa.

Authors:  M F Jonkman; M C de Jong; K Heeres; H H Pas; J B van der Meer; K Owaribe; A M Martinez de Velasco; C M Niessen; A Sonnenberg
Journal:  J Clin Invest       Date:  1995-03       Impact factor: 14.808

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