Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure
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...
| Main Authors: | , , , , , , , , , , , , , , |
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| Format: | Article |
| Language: | English |
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eLife Sciences Publications Ltd
2018-06-01
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| Series: | eLife |
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| Online Access: | https://elifesciences.org/articles/37131 |
| _version_ | 1811200772421976064 |
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| author | 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 |
| author_facet | 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 |
| author_sort | Ian A Swinburne |
| collection | DOAJ |
| description | 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. |
| first_indexed | 2024-04-12T02:09:17Z |
| format | Article |
| id | doaj.art-9605396941f44e5497b36a7a18e10d89 |
| institution | Directory Open Access Journal |
| issn | 2050-084X |
| language | English |
| last_indexed | 2024-04-12T02:09:17Z |
| publishDate | 2018-06-01 |
| publisher | eLife Sciences Publications Ltd |
| record_format | Article |
| series | eLife |
| spelling | doaj.art-9605396941f44e5497b36a7a18e10d892022-12-22T03:52:27ZengeLife Sciences Publications LtdeLife2050-084X2018-06-01710.7554/eLife.37131Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressureIan A Swinburne0https://orcid.org/0000-0003-4162-0508Kishore R Mosaliganti1Srigokul Upadhyayula2https://orcid.org/0000-0002-6911-0270Tsung-Li Liu3David G C Hildebrand4https://orcid.org/0000-0002-2079-3517Tony Y -C Tsai5Anzhi Chen6Ebaa Al-Obeidi7Anna K Fass8Samir Malhotra9Florian Engert10Jeff W Lichtman11https://orcid.org/0000-0002-0208-3212Tomas Kirchhausen12Eric Betzig13Sean G Megason14https://orcid.org/0000-0002-9330-2934Department of Systems Biology, Harvard Medical School, Boston, United StatesDepartment of Systems Biology, Harvard Medical School, Boston, United StatesDepartment of Pediatrics, Harvard Medical School, Boston, United States; Program in Cellular and Molecular Medicine, Boston Children’s Hospital, Boston, United States; Janelia Research Campus, Howard Hughes Medical Institute, Ashburn, United StatesJanelia Research Campus, Howard Hughes Medical Institute, Ashburn, United StatesDepartment of Molecular and Cellular Biology, Harvard University, Cambridge, United StatesDepartment of Systems Biology, Harvard Medical School, Boston, United StatesDepartment of Systems Biology, Harvard Medical School, Boston, United StatesDepartment of Systems Biology, Harvard Medical School, Boston, United StatesDepartment of Systems Biology, Harvard Medical School, Boston, United StatesDepartment of Systems Biology, Harvard Medical School, Boston, United StatesDepartment of Molecular and Cellular Biology, Harvard University, Cambridge, United StatesDepartment of Molecular and Cellular Biology, Harvard University, Cambridge, United StatesDepartment of Pediatrics, Harvard Medical School, Boston, United States; Program in Cellular and Molecular Medicine, Boston Children’s Hospital, Boston, United States; Department of Cell Biology, Harvard Medical School, Boston, United StatesJanelia Research Campus, Howard Hughes Medical Institute, Ashburn, United StatesDepartment of Systems Biology, Harvard Medical School, Boston, United StatesThe 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.https://elifesciences.org/articles/37131inner earpressuretimelapselamellavalve |
| spellingShingle | 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 Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure eLife inner ear pressure timelapse lamella valve |
| title | Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure |
| title_full | Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure |
| title_fullStr | Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure |
| title_full_unstemmed | Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure |
| title_short | Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure |
| title_sort | lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure |
| topic | inner ear pressure timelapse lamella valve |
| url | https://elifesciences.org/articles/37131 |
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