Literature DB >> 9856843

Novel and de novo glycine substitution mutations in the type VII collagen gene (COL7A1) in dystrophic epidermolysis bullosa: implications for genetic counseling.

F Rouan1, L Pulkkinen, M F Jonkman, J W Bauer, P B Cserhalmi-Friedman, A M Christiano, J Uitto.   

Abstract

The dystrophic forms of epidermolysis bullosa (DEB) are due to mutations in the type VII collagen gene (COL7A1). In dominant DEB, a characteristic genetic lesion is a glycine substitution mutation within the collagenous domain of the protein. In this study, we have examined the molecular basis of six new families in which the proband has clinical features and/or ultrastructural findings consistent with DEB. The results revealed a glycine substitution mutation in all six families, four of which are novel and previously unpublished. In three families with clinically unaffected parents, de novo mutations G2043R and G2040V were found. These results emphasize the predominance of glycine substitution mutations in dominant DEB, and indicate that in some cases the phenotype is due to de novo dominant mutations.

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Year:  1998        PMID: 9856843     DOI: 10.1046/j.1523-1747.1998.00422.x

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


  1 in total

1.  Genetic studies of 20 Japanese families of dystrophic epidermolysis bullosa.

Authors:  Daisuke Sawamura; Maki Goto; Kana Yasukawa; Kazuko Sato-Matsumura; Hideki Nakamura; Kei Ito; Hiroyuki Nakamura; Yuki Tomita; Hiroshi Shimizu
Journal:  J Hum Genet       Date:  2005-09-28       Impact factor: 3.172

  1 in total

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