PUBLICATION

In Vivo imaging of molecular interactions at damaged sarcolemma

Authors
Roostalu, U., and Strähle, U.
ID
ZDB-PUB-120326-2
Date
2012
Source
Developmental Cell   22(3): 515-529 (Journal)
Registered Authors
Roostalu, Urmas, Strähle, Uwe
Keywords
none
MeSH Terms
  • Amino Acid Sequence
  • Animals
  • Annexins/genetics
  • Annexins/physiology
  • Base Sequence
  • Disease Models, Animal
  • Membrane Proteins/genetics
  • Molecular Sequence Data
  • Muscular Diseases/genetics
  • Muscular Diseases/physiopathology*
  • Muscular Dystrophies, Limb-Girdle/genetics
  • Muscular Dystrophies, Limb-Girdle/physiopathology*
  • Sarcolemma/genetics
  • Sarcolemma/physiology*
  • Sarcolemma/ultrastructure
  • Zebrafish/genetics
  • Zebrafish/physiology
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/physiology
PubMed
22421042 Full text @ Dev. Cell
Abstract

Muscle cells have a remarkable capability to repair plasma membrane lesions. Mutations in dysferlin (dysf) are known to elicit a progressive myopathy in humans, probably due to impaired sarcolemmal repair. We show here that loss of Dysf and annexin A6 (Anxa6) function lead to myopathy in zebrafish. By use of high-resolution imaging of myofibers in intact animals, we reveal sequential phases in sarcolemmal repair. Initially, membrane vesicles enriched in Dysf together with cytoplasmic Anxa6 form a tight patch at the lesion independently of one another. In the subsequent steps, annexin A2a (Anxa2a) followed by annexin A1a (Anxa1a) accumulate at the patch; the recruitment of these annexins depends on Dysf and Anxa6. Thus, sarcolemmal repair relies on the ordered assembly of a protein-membrane scaffold. Moreover, we provide several lines of evidence that the membrane for sarcolemmal repair is derived from a specialized plasma membrane compartment.

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Human Disease / Model
Sequence Targeting Reagents
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Mapping