Mutations in the dystrophin-like dys-1 gene of Caenorhabditis elegans result in reduced acetylcholinesterase activity

Mutations of the Caenorhabditis elegans dystrophin/utrophin-like dys-1 gene lead to hyperactivity and hypercontraction of the animals. In addition dys -1 mutants are hypersensitive to acetylcholine and acetylcholinesterase inh ibitors. We investigated this phenotype further by assaying acetylcholinest erase activity, Total extracts from three different dys-1 alleles showed si gnificantly less acetylcholinesterase-specific activity than wild-type cont rols. In addition. double mutants carrying a mutation in the dys-1 gene plu s a mutation in either of the two major acetylcholinesterase genes (ace-1 a nd ace-2) display locomotor defects consistent with a strong reduction of a cetylcholinesterases, whereas none of the single mutants does, Therefore, i n C. elegans, disruption of the dystrophin/utrophin-like dys-1 gene affects acetylcholinesterase activity. (C) 1999 Federation of European Biochemical Societies.