Genetic modifier screens reveal new components that interact with the Drosophila dystroglycan-dystrophin complex

PLoS One. 2008 Jun 11;3(6):e2418. doi: 10.1371/journal.pone.0002418.

Abstract

The Dystroglycan-Dystrophin (Dg-Dys) complex has a capacity to transmit information from the extracellular matrix to the cytoskeleton inside the cell. It is proposed that this interaction is under tight regulation; however the signaling/regulatory components of Dg-Dys complex remain elusive. Understanding the regulation of the complex is critical since defects in this complex cause muscular dystrophy in humans. To reveal new regulators of the Dg-Dys complex, we used a model organism Drosophila melanogaster and performed genetic interaction screens to identify modifiers of Dg and Dys mutants in Drosophila wing veins. These mutant screens revealed that the Dg-Dys complex interacts with genes involved in muscle function and components of Notch, TGF-beta and EGFR signaling pathways. In addition, components of pathways that are required for cellular and/or axonal migration through cytoskeletal regulation, such as Semaphorin-Plexin, Frazzled-Netrin and Slit-Robo pathways show interactions with Dys and/or Dg. These data suggest that the Dg-Dys complex and the other pathways regulating extracellular information transfer to the cytoskeletal dynamics are more intercalated than previously thought.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Drosophila Proteins / metabolism
  • Drosophila melanogaster / genetics*
  • Drosophila melanogaster / metabolism
  • Dystroglycans / metabolism*
  • Dystrophin / metabolism*
  • ErbB Receptors / metabolism
  • Mutation
  • Receptors, Notch / metabolism
  • Signal Transduction
  • Transforming Growth Factor beta / metabolism

Substances

  • Drosophila Proteins
  • Dystrophin
  • N protein, Drosophila
  • Receptors, Notch
  • Transforming Growth Factor beta
  • Dystroglycans
  • ErbB Receptors