ArticleThe Journal of cell biology2004
Characterization of a hypercontraction-induced myopathy in Drosophila caused by mutations in Mhc.
Article in The Journal of cell biology, 2004. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.
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Who cites it
15 citing papers in PubMed, 34 citations in OpenAlex.
- Drosophila Transglutaminase preserves the integrity of muscle attachments with and without mechanical strain.Journal of cell science · 2026Article
- Titin-dependent biomechanical feedback tailors sarcomeres to specialized muscle functions in insects.Science advances · 2025Article
- Filamin protects myofibrils from contractile damage through changes in its mechanosensory region.PLoS genetics · 2024Article
- Loss of the extracellular matrix protein Perlecan disrupts axonal and synaptic stability duringeLife · 2023Article
- Bidirectional regulation of glial potassium buffering - glioprotection versus neuroprotection.eLife · 2021Article
- Reductions in ATPase activity, actin sliding velocity, and myofibril stability yield muscle dysfunction in Drosophila models of myosin-based Freeman-Sheldon syndrome.Molecular biology of the cell · 2019Article
- A tissue communication network coordinating innate immune response during muscle stress.Journal of cell science · 2018Article
- In vivo quantitative analysis of Talin turnover in response to force.Molecular biology of the cell · 2015Article
- Elevated expression of the integrin-associated protein PINCH suppresses the defects of Drosophila melanogaster muscle hypercontraction mutants.PLoS genetics · 2013Article
- Mechanical force regulates integrin turnover in Drosophila in vivo.Nature cell biology · 2012Article
- Alternative relay and converter domains tune native muscle myosin isoform function in Drosophila.Journal of molecular biology · 2012Article
- Hyperthermic seizures and aberrant cellular homeostasis in Drosophila dystrophic muscles.Scientific reports · 2011Article
- Myosin transducer mutations differentially affect motor function, myofibril structure, and the performance of skeletal and cardiac muscles.Molecular biology of the cell · 2008Article
- Other model organisms for sarcomeric muscle diseases.Advances in experimental medicine and biology · 2008Review
- The Drosophila ARC homolog regulates behavioral responses to starvation.Molecular and cellular neurosciences · 2007Article
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Authors and funding
2 authors at 1 institution in 1 country.
Funding
Abstract
The Myosin heavy chain (Mhc) locus encodes the muscle-specific motor mediating contraction in Drosophila. In a screen for temperature-sensitive behavioral mutants, we have identified two dominant Mhc alleles that lead to a hypercontraction-induced myopathy. These mutants are caused by single point mutations in the ATP binding/hydrolysis domain of Mhc and lead to degeneration of the flight muscles. Electrophysiological analysis in the adult giant fiber flight circuit demonstrates temperature-dependent seizure activity that requires neuronal input, as genetic blockage of neuronal activity suppresses the electrophysiological seizure defects. Intracellular recordings at the third instar neuromuscular junction show spontaneous muscle movements in the absence of neuronal stimulation and extracellular Ca2+, suggesting a dysregulation of intracellular calcium homeostasis within the muscle or an alteration of the Ca2+ dependence of contraction. Characterization of these new Mhc alleles suggests that hypercontraction occurs via a mechanism, which is molecularly distinct from mutants identified previously in troponin I and troponin T.
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