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
  • Muscle Fibers, Skeletal/metabolism
  • Muscle Fibers, Skeletal/ultrastructure
  • Muscle, Skeletal/metabolism*
  • Muscle, Skeletal/ultrastructure
  • Cell Line
  • Adaptor Proteins, Signal Transducing/metabolism*
  • Muscle Proteins/metabolism*
  • Nerve Tissue Proteins/metabolism*
  • Caveolae/metabolism
  • Protein Binding
  • Tumor Suppressor Proteins/metabolism*
  • Animals
  • Sarcolemma/metabolism*
  • Sarcolemma/ultrastructure
  • Zebrafish Proteins/metabolism*
(all 20)
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
Figures
Figure Gallery (4 images)
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rpstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rpstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rpstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rp; pc10Tgstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rp; pc10Tgstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rp; pc10Tgstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rp; uq11rpTgstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rp; uq11rpTgstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rp; uq11rpTgstandard conditionsDay 5
cavin4buq6rp/uq6rp; cavin4auq5rp/uq5rp; uq16rpTgstandard conditionsProtruding-mouth
1 - 10 of 10
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Mutations / Transgenics
Allele Construct Type Affected Genomic Region
pc10TgTransgenic Insertion
    uq11rpTgTransgenic Insertion
      uq5rp
        Insertion
        uq6rp
          Point Mutation
          uq16rpTgTransgenic Insertion
            uq17rpTgTransgenic Insertion
              uq18rpTgTransgenic Insertion
                uq19rpTgTransgenic Insertion
                  1 - 8 of 8
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                  Human Disease / Model
                  No data available
                  Sequence Targeting Reagents
                  Target Reagent Reagent Type
                  cavin4aCRISPR1-cavin4aCRISPR
                  1 - 1 of 1
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                  Fish
                  Antibodies
                  Name Type Antigen Genes Isotypes Host Organism
                  Ab4-dag1monoclonalIgG1Mouse
                  1 - 1 of 1
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                  Orthology
                  No data available
                  Engineered Foreign Genes
                  Marker Marker Type Name
                  EGFPEFGEGFP
                  GCaMPEFGGCaMP
                  GFPEFGGFP
                  mCloverEFGmClover
                  1 - 4 of 4
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                  Mapping
                  No data available