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Hoffmann, R. A wiki for the life sciences where authorship matters. Nature Genetics (2008)
 
 
 
 
 

Dp71 can restore the dystrophin-associated glycoprotein complex in muscle but fails to prevent dystrophy.

Two lines of transgenic mdx mice have been generated that express a 71 kD non-muscle isoform of dystrophin ( Dp71) in skeletal muscle. This isoform contains the cysteine-rich and C-terminal domains of dystrophin, but lacks the N-terminal actin-binding and central spectrin-like repeat domains. Dp71 was associated with the sarcolemma membrane, where it restored normal expression and localization of all members of the dystrophin-associated glycoprotein complex. However, the skeletal muscle pathology of the transgenic mdx mice remained severe. These results indicate that the dystrophin C terminus cannot function independently to prevent dystrophic symptoms and confirms predictions based on patient data that both the N and C-terminal domains are required for normal dystrophin function.[1]

References

  1. Dp71 can restore the dystrophin-associated glycoprotein complex in muscle but fails to prevent dystrophy. Cox, G.A., Sunada, Y., Campbell, K.P., Chamberlain, J.S. Nat. Genet. (1994) [Pubmed]
 
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