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Disruption of ERBB2IP is not associated with dystrophic epidermolysis bullosa in both father and son carrying a balanced 5;13 translocation.
- Source :
-
The Journal of investigative dermatology [J Invest Dermatol] 2005 Oct; Vol. 125 (4), pp. 700-4. - Publication Year :
- 2005
-
Abstract
- Mutations in the type VII collagen gene (COL7A1) cause autosomal recessive and autosomal dominant inherited dystrophic epidermolysis bullosa (DEB). We report a family with three individuals who present blistering, scarring, hypo- and hyperpigmentation, and nail dystrophy suggestive for DEB. Whereas father and son carry a 5;13 translocation, the daughter shows a normal karyotype. Segregation analysis revealed that all affected family members inherited the same COL7A1 allele. Mutation analysis disclosed a heterozygous missense mutation, c.6227G > A (p.G2076D), in COL7A1 in all affected individuals. Delineation of the translocation breakpoints showed that the ERBB2IP (erbb2 interacting protein or Erbin) gene is disrupted in 5q13.1 and GPC6 in 13q32. GPC6 encodes glypican 6 belonging to a family of cell surface heparan sulfate proteoglycans. The binding partners of Erbin, BP230 (BPAG1) and the integrin beta4 subunit, both involved in hemidesmosome (HD) function, and the presence of Erbin in HD suggested that it plays a role in establishment and maintenance of cell-basement membrane adhesions. However, loss of function of one ERBB2IP copy or expression of a putative novel ERBB2IP fusion protein did not apparently modulate the DEB phenotype in both translocation patients. Nonetheless, one cannot yet exclude that ERBB2IP is a candidate for human blistering disorders such as epidermolysis bullosa.
- Subjects :
- Adaptor Proteins, Signal Transducing
Adult
Child
Chromosomes, Human, Pair 13
Chromosomes, Human, Pair 5
Fathers
Female
Glypicans
Heparan Sulfate Proteoglycans genetics
Humans
Male
Middle Aged
Nuclear Family
Carrier Proteins genetics
Collagen Type VII genetics
Epidermolysis Bullosa Dystrophica genetics
Translocation, Genetic
Subjects
Details
- Language :
- English
- ISSN :
- 0022-202X
- Volume :
- 125
- Issue :
- 4
- Database :
- MEDLINE
- Journal :
- The Journal of investigative dermatology
- Publication Type :
- Academic Journal
- Accession number :
- 16185268
- Full Text :
- https://doi.org/10.1111/j.0022-202X.2005.23875.x