Journal
BIOESSAYS
Volume 33, Issue 12, Pages 956-965Publisher
WILEY
DOI: 10.1002/bies.201100082
Keywords
C; elegans; Drosophila; motor neuron; neuromuscular disease; survival motor neuron
Categories
Funding
- Functional Genomics Unit of the Medical Research Council of the UK
- SMA Europe
- MRC [MC_U137788471] Funding Source: UKRI
- Medical Research Council [MC_U137788471] Funding Source: researchfish
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Invertebrate genetic models with their tractable neuromuscular systems are effective vehicles for the study of human nerve and muscle disorders. This is exemplified by insights made into spinal muscular atrophy (SMA) using the fruit fly Drosophila melanogaster and the nematode worm Caenorhabditis elegans. For speed and economy, these invertebrates offer convenient, whole-organism platforms for genetic screening as well as RNA interference (RNAi) and chemical library screens, permitting the rapid testing of hypotheses related to disease mechanisms and the exploration of new therapeutic routes and drug candidates. Here, we discuss recent developments encompassing synaptic physiology, RNA processing, and screening of compound and genome-scale RNAi libraries, showcasing the importance of invertebrate SMA models.
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