PUBLICATION
Cavin4 interacts with Bin1 to promote T-tubule formation and stability in developing skeletal muscle
- Authors
- Lo, H.P., Lim, Y.W., Xiong, Z., Martel, N., Ferguson, C., Ariotti, N., Giacomotto, J., Rae, J., Floetenmeyer, M., Moradi, S.V., Gao, Y., Tillu, V.A., Xia, D., Wang, H., Rahnama, S., Nixon, S.J., Bastiani, M., Day, R.D., Smith, K.A., Palpant, N.J., Johnston, W.A., Alexandrov, K., Collins, B.M., Hall, T.E., Parton, R.G.
- ID
- ZDB-PUB-211012-14
- Date
- 2021
- Source
- The Journal of cell biology 220(12): (Journal)
- Registered Authors
- Giacomotto, Jean, Hall, Thomas, Lo, Harriet, Parton, Robert G., Smith, Kelly
- Keywords
- none
- MeSH Terms
-
- Imaging, Three-Dimensional
- Embryo, Nonmammalian/metabolism
- Zebrafish/embryology
- Zebrafish/metabolism*
- Mice, Inbred C57BL
- PubMed
- 34633413 Full text @ J. Cell Biol.
Abstract
The cavin proteins are essential for caveola biogenesis and function. Here, we identify a role for the muscle-specific component, Cavin4, in skeletal muscle T-tubule development by analyzing two vertebrate systems, mouse and zebrafish. In both models, Cavin4 localized to T-tubules, and loss of Cavin4 resulted in aberrant T-tubule maturation. In zebrafish, which possess duplicated cavin4 paralogs, Cavin4b was shown to directly interact with the T-tubule-associated BAR domain protein Bin1. Loss of both Cavin4a and Cavin4b caused aberrant accumulation of interconnected caveolae within the T-tubules, a fragmented T-tubule network enriched in Caveolin-3, and an impaired Ca2+ response upon mechanical stimulation. We propose a role for Cavin4 in remodeling the T-tubule membrane early in development by recycling caveolar components from the T-tubule to the sarcolemma. This generates a stable T-tubule domain lacking caveolae that is essential for T-tubule function.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping