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It is caused by a loss-of-function mutation in the survival motor neuron (<jats:italic>SMN1<\/jats:italic>) gene.<jats:italic>Caenorhabditis elegans<\/jats:italic>mutants lacking<jats:italic>SMN<\/jats:italic>recapitulate several aspects of the disease including impaired movement and shorted life span. We examined whether genes previously implicated in life span extension conferred benefits to<jats:italic>C. elegans<\/jats:italic>lacking SMN. We find that reducing daf-2\/insulin receptor signaling activity promotes survival and improves locomotor behavior in this<jats:italic>C. elegans<\/jats:italic>model of SMA. The locomotor dysfunction in<jats:italic>C. elegans<\/jats:italic>lacking<jats:italic>SMN<\/jats:italic>correlated with structural and functional abnormalities in GABAergic neuromuscular junctions (NMJs). Moreover, we demonstrated that reduction in<jats:italic>daf-2<\/jats:italic>signaling reversed these abnormalities. Remarkably, enhancing GABAergic neurotransmission alone was able to correct the locomotor dysfunction. Our work indicated that an imbalance of excitatory\/inhibitory activity within motor circuits and underlies motor system dysfunction in this SMA model. Interventions aimed at restoring the balance of excitatory\/inhibitory activity in motor circuits could be of benefit to individuals with SMA.<\/jats:p>","DOI":"10.1523\/eneuro.0289-18.2018","type":"journal-article","created":{"date-parts":[[2018,11,16]],"date-time":"2018-11-16T18:00:33Z","timestamp":1542391233000},"page":"ENEURO.0289-18.2018","source":"Crossref","is-referenced-by-count":12,"title":["Enhancing GABAergic Transmission Improves Locomotion in a<i>Caenorhabditis elegans<\/i>Model of Spinal Muscular Atrophy"],"prefix":"10.1523","volume":"5","author":[{"given":"Chia-Yen","family":"Wu","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David A.","family":"Gagnon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juliette 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