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Reductions in ATPase activity, actin sliding velocity, and myofibril stability yield muscle dysfunction in Drosophila models of myosin-based Freeman–Sheldon syndrome
Using Drosophila melanogaster, we created the first animal models for myosin-based Freeman–Sheldon syndrome (FSS), a dominant form of distal arthrogryposis defined by congenital facial and distal skeletal muscle contractures. Electron microscopy of homozygous mutant indirect flight muscles showed no...
Autores principales: | Rao, Deepti S., Kronert, William A., Guo, Yiming, Hsu, Karen H., Sarsoza, Floyd, Bernstein, Sanford I. |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
The American Society for Cell Biology
2019
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6337914/ https://www.ncbi.nlm.nih.gov/pubmed/30379605 http://dx.doi.org/10.1091/mbc.E18-08-0526 |
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