PUBLICATION
Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure
- Authors
- Swinburne, I.A., Mosaliganti, K.R., Upadhyayula, S., Liu, T.L., Hildebrand, D.G.C., Tsai, T.Y., Chen, A., Al-Obeidi, E., Fass, A.K., Malhotra, S., Engert, F., Lichtman, J.W., Kirchausen, T., Betzig, E., Megason, S.G.
- ID
- ZDB-PUB-180622-24
- Date
- 2018
- Source
- eLIFE 7: (Journal)
- Registered Authors
- Chen, Anzhi, Engert, Florian, Megason, Sean, Mosaliganti, Kishore, Swinburne, Ian
- Keywords
- cell biology, developmental biology, inner ear, lamella, pressure, stem cells, timelapse, valve, zebrafish
- MeSH Terms
-
- Animals
- Animals, Genetically Modified
- Embryo, Nonmammalian
- Endolymphatic Sac/anatomy & histology
- Endolymphatic Sac/physiology
- Endolymphatic Sac/ultrastructure*
- Female
- Gene Expression
- Hearing/physiology*
- Homeostasis/physiology
- Hydrostatic Pressure
- In Situ Hybridization, Fluorescence
- Larva/anatomy & histology
- Larva/physiology
- Larva/ultrastructure*
- Male
- Microscopy, Electron
- Mutation
- Time-Lapse Imaging
- Transcription Factors/genetics*
- Transcription Factors/metabolism
- Zebrafish
- Zebrafish Proteins/genetics*
- Zebrafish Proteins/metabolism
- PubMed
- 29916365 Full text @ Elife
Citation
Swinburne, I.A., Mosaliganti, K.R., Upadhyayula, S., Liu, T.L., Hildebrand, D.G.C., Tsai, T.Y., Chen, A., Al-Obeidi, E., Fass, A.K., Malhotra, S., Engert, F., Lichtman, J.W., Kirchausen, T., Betzig, E., Megason, S.G. (2018) Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure. eLIFE. 7.
Abstract
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.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping