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Loss of dystrophin and the microtubule-binding protein ELP-1 causes progressive paralysis and death of adult C. elegans
Hueston, Jennifer L. Suprenant, Kathy A.
Hueston, Jennifer L.
Suprenant, Kathy A.
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Abstract
EMAP-like proteins (ELPs) are conserved microtubule-binding proteins that function during cell division and in the behavior of post-mitotic cells. In C. elegans, ELP-1 is broadly expressed in many cells and tissues including the touch receptor neurons and body wall muscle. Within muscle, ELP-1 is associated with a microtubule network that is closely opposed to the integrin-based adhesion sites called dense bodies. To examine ELP-1 function we utilized an elp-1 RNA interference assay and screened for synthetic interactions with mutated adhesion site proteins. We reveal a synthetic lethal relationship between ELP-1 and the dystrophin-like protein, DYS-1. Reduction of ELP-1 in a dystrophin [dys-1(cx18)] mutant results in adult animals with motility defects, splayed and hypercontracted muscle with altered cholinergic signaling. Worms fill with vesicles, become flaccid and die. We conclude that ELP-1 is a genetic modifier of a C. elegans model of muscular dystrophy.
Description
This is the peer reviewed version of the following article: Hueston, J. L. and Suprenant, K. A. (2009), Loss of dystrophin and the microtubule-binding protein ELP-1 causes progressive paralysis and death of adult C. elegans. Dev. Dyn., 238: 1878–1886. doi:10.1002/dvdy.22007, which has been published in final form at http://doi.org/10.1002/dvdy.22007. This article may be used for non-commercial purposes in accordance with Wiley Terms and Conditions for Self-Archiving.
Date
2009-08
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Wiley
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Keywords
Muscle physiology, Cytoskeleton, Cell adhesion, Cholinergic agents, Muscular dystrophy
Citation
Hueston, J. L. and Suprenant, K. A. (2009), Loss of dystrophin and the microtubule-binding protein ELP-1 causes progressive paralysis and death of adult C. elegans. Dev. Dyn., 238: 1878–1886. doi:10.1002/dvdy.22007