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...

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Detalles Bibliográficos
Autores principales: Rao, Deepti S., Kronert, William A., Guo, Yiming, Hsu, Karen H., Sarsoza, Floyd, Bernstein, Sanford I.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The American Society for Cell Biology 2019
Materias:
Articles
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

Internet

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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