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Gene Review

dys-1  -  Protein DYS-1

Caenorhabditis elegans

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Disease relevance of dystrophin


High impact information on dystrophin


Chemical compound and disease context of dystrophin


Biological context of dystrophin

  • Dystrobrevins are dystrophin-associated proteins potentially involved in signal transduction [2].
  • Phenotypes similar to that of dys-1 were obtained, both in the wild-type background and in combination with other mutations [7].
  • Using RNA interference and existing mutants, we genetically impaired the excitation-contraction cascade at specific points in a dystrophin-deficient C. elegans strain which normally undergoes extensive muscle degeneration [9].
  • Based on reporter gene analysis and heterologous promoter expression, the site of action of the dys-1 gene seems to be in muscles [10].
  • A chimeric transgene in which the C-terminal end of the protein has been replaced by the human dystrophin sequence is able to partly suppress the phenotype of the dys-1 mutants, showing that both proteins share some functional similarity [10].

Anatomical context of dystrophin


Associations of dystrophin with chemical compounds


Physical interactions of dystrophin


Other interactions of dystrophin

  • These findings suggest that: (1) dystrobrevin and dystrophin are functionally related and (2) their disruption impairs cholinergic signalling [14].
  • Therefore, in C. elegans, disruption of the dystrophin/utrophin-like dys-1 gene affects acetylcholinesterase activity [15].
  • In addition, double mutants carrying a mutation in the dys-1 gene plus a mutation in either of the two major acetylcholinesterase genes (ace-1 and ace-2) display locomotor defects consistent with a strong reduction of acetylcholinesterases, whereas none of the single mutants does [15].

Analytical, diagnostic and therapeutic context of dystrophin


  1. Molecular, genetic and physiological characterisation of dystrobrevin-like (dyb-1) mutants of Caenorhabditis elegans. Gieseler, K., Mariol, M.C., Bessou, C., Migaud, M., Franks, C.J., Holden-Dye, L., Ségalat, L. J. Mol. Biol. (2001) [Pubmed]
  2. Overexpression of dystrobrevin delays locomotion defects and muscle degeneration in a dystrophin-deficient Caenorhabditis elegans. Gieseler, K., Grisoni, K., Mariol, M.C., Ségalat, L. Neuromuscul. Disord. (2002) [Pubmed]
  3. Defective membrane repair in dysferlin-deficient muscular dystrophy. Bansal, D., Miyake, K., Vogel, S.S., Groh, S., Chen, C.C., Williamson, R., McNeil, P.L., Campbell, K.P. Nature (2003) [Pubmed]
  4. Muscular degeneration in the absence of dystrophin is a calcium-dependent process. Mariol, M.C., Ségalat, L. Curr. Biol. (2001) [Pubmed]
  5. The SLO-1 BK channel of Caenorhabditis elegans is critical for muscle function and is involved in dystrophin-dependent muscle dystrophy. Carre-Pierrat, M., Grisoni, K., Gieseler, K., Mariol, M.C., Martin, E., Jospin, M., Allard, B., Ségalat, L. J. Mol. Biol. (2006) [Pubmed]
  6. Gene expression profiling studies on Caenorhabditis elegans dystrophin mutants dys-1(cx-35) and dys-1(cx18). Towers, P.R., Lescure, P., Baban, D., Malek, J.A., Duarte, J., Jones, E., Davies, K.E., S??galat, L., Sattelle, D.B. Genomics (2006) [Pubmed]
  7. Genetic evidence for a dystrophin-glycoprotein complex (DGC) in Caenorhabditis elegans. Grisoni, K., Martin, E., Gieseler, K., Mariol, M.C., Ségalat, L. Gene (2002) [Pubmed]
  8. Blocking of Striated Muscle Degeneration by Serotonin in C. elegans. Carre-Pierrat, M., Mariol, M.C., Chambonnier, L., Laugraud, A., Heskia, F., Giacomotto, J., Ségalat, L. J. Muscle Res. Cell. Motil. (2006) [Pubmed]
  9. Dystrophin-dependent muscle degeneration requires a fully functional contractile machinery to occur in C. elegans. Mariol, M.C., Martin, E., Chambonnier, L., Ségalat, L. Neuromuscul. Disord. (2007) [Pubmed]
  10. Mutations in the Caenorhabditis elegans dystrophin-like gene dys-1 lead to hyperactivity and suggest a link with cholinergic transmission. Bessou, C., Giugia, J.B., Franks, C.J., Holden-Dye, L., Ségalat, L. Neurogenetics (1998) [Pubmed]
  11. Prednisone in dystrophin-deficient Caernorabditis elegans. De Luca, A. Neuromuscul. Disord. (2004) [Pubmed]
  12. SNF-6 is an acetylcholine transporter interacting with the dystrophin complex in Caenorhabditis elegans. Kim, H., Rogers, M.J., Richmond, J.E., McIntire, S.L. Nature (2004) [Pubmed]
  13. Dystrobrevin requires a dystrophin-binding domain to function in Caenorhabditis elegans. Grisoni, K., Gieseler, K., Ségalat, L. Eur. J. Biochem. (2002) [Pubmed]
  14. Dystrobrevin- and dystrophin-like mutants display similar phenotypes in the nematode Caenorhabditis elegans. Gieseler, K., Bessou, C., Ségalat, L. Neurogenetics (1999) [Pubmed]
  15. Mutations in the dystrophin-like dys-1 gene of Caenorhabditis elegans result in reduced acetylcholinesterase activity. Giugia, J., Gieseler, K., Arpagaus, M., Ségalat, L. FEBS Lett. (1999) [Pubmed]
  16. Prednisone reduces muscle degeneration in dystrophin-deficient Caenorhabditis elegans. Gaud, A., Simon, J.M., Witzel, T., Carre-Pierrat, M., Wermuth, C.G., Ségalat, L. Neuromuscul. Disord. (2004) [Pubmed]
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