RRC ID 15355
Author Marrone AK, Kucherenko MM, Rishko VM, Shcherbata HR.
Title New dystrophin/dystroglycan interactors control neuron behavior in Drosophila eye.
Journal BMC Neurosci
Abstract BACKGROUND:The Dystrophin Glycoprotein Complex (DGC) is a large multi-component complex that is well known for its function in muscle tissue. When the main components of the DGC, Dystrophin (Dys) and Dystroglycan (Dg) are affected cognitive impairment and mental retardation in addition to muscle degeneration can occur. Previously we performed an array of genetic screens using a Drosophila model for muscular dystrophy in order to find novel DGC interactors aiming to elucidate the signaling role(s) in which the complex is involved. Since the function of the DGC in the brain and nervous system has not been fully defined, we have here continued to analyze the DGC modifiers' function in the developing Drosophila brain and eye.
RESULTS:Given that disruption of Dys and Dg leads to improper photoreceptor axon projections into the lamina and eye neuron elongation defects during development, we have determined the function of previously screened components and their genetic interaction with the DGC in this tissue. Our study first found that mutations in chif, CG34400, Nrk, Lis1, capt and Cam cause improper axon path-finding and loss of SP2353, Grh, Nrk, capt, CG34400, vimar, Lis1 and Cam cause shortened rhabdomere lengths. We determined that Nrk, mbl, capt and Cam genetically interact with Dys and/or Dg in these processes. It is notable that most of the neuronal DGC interacting components encountered are involved in regulation of actin dynamics.
CONCLUSIONS:Our data indicate possible DGC involvement in the process of cytoskeletal remodeling in neurons. The identification of new components that interact with the DGC not only helps to dissect the mechanism of axon guidance and eye neuron differentiation but also provides a great opportunity for understanding the signaling mechanisms by which the cell surface receptor Dg communicates via Dys with the actin cytoskeleton.
Volume 12
Pages 93
Published 2011-9-26
DOI 10.1186/1471-2202-12-93
PII 1471-2202-12-93
PMID 21943192
PMC PMC3217851
MeSH Animals Axons / metabolism* Axons / pathology Axons / physiology Drosophila Proteins / genetics Drosophila Proteins / metabolism* Drosophila Proteins / physiology Drosophila melanogaster Dystroglycans / genetics Dystroglycans / metabolism* Dystroglycans / physiology Dystrophin / genetics Dystrophin / metabolism* Dystrophin / physiology Gene Expression Regulation, Developmental / physiology Muscular Dystrophies / genetics Muscular Dystrophies / metabolism Muscular Dystrophies / pathology Mutation Photoreceptor Cells, Invertebrate / metabolism* Photoreceptor Cells, Invertebrate / pathology Photoreceptor Cells, Invertebrate / physiology Signal Transduction / genetics Signal Transduction / physiology
IF 2.811
Times Cited 21
Drosophila DGRC#111319