eLife (Jun 2018)

Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure

  • Ian A Swinburne,
  • Kishore R Mosaliganti,
  • Srigokul Upadhyayula,
  • Tsung-Li Liu,
  • David G C Hildebrand,
  • Tony Y -C Tsai,
  • Anzhi Chen,
  • Ebaa Al-Obeidi,
  • Anna K Fass,
  • Samir Malhotra,
  • Florian Engert,
  • Jeff W Lichtman,
  • Tomas Kirchhausen,
  • Eric Betzig,
  • Sean G Megason

DOI
https://doi.org/10.7554/eLife.37131
Journal volume & issue
Vol. 7

Abstract

Read online

The inner ear is a fluid-filled closed-epithelial structure whose function requires maintenance of an internal hydrostatic pressure and fluid composition. The endolymphatic sac (ES) is a dead-end epithelial tube connected to the inner ear whose function is unclear. ES defects can cause distended ear tissue, a pathology often seen in hearing and balance disorders. Using live imaging of zebrafish larvae, we reveal that the ES undergoes cycles of slow pressure-driven inflation followed by rapid deflation. Absence of these cycles in lmx1bb mutants leads to distended ear tissue. Using serial-section electron microscopy and adaptive optics lattice light-sheet microscopy, we find a pressure relief valve in the ES comprised of partially separated apical junctions and dynamic overlapping basal lamellae that separate under pressure to release fluid. We propose that this lmx1-dependent pressure relief valve is required to maintain fluid homeostasis in the inner ear and other fluid-filled cavities.

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