@article{3962f3f78e5e44a4b05c5e6d500fcd09,
title = "Lamellar projections in the endolymphatic sac act as a relief valve to regulate inner ear pressure",
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.",
author = "Swinburne, {Ian A.} and Mosaliganti, {Kishore R.} and Srigokul Upadhyayula and Liu, {Tsung Li} and Hildebrand, {David G.C.} and Tsai, {Tony Y.C.} and Anzhi Chen and Ebaa Al-Obeidi and Fass, {Anna K.} and Samir Malhotra and Florian Engert and Lichtman, {Jeff W.} and Tomas Kirchhausen and Eric Betzig and Megason, {Sean G.}",
note = "Funding Information: We thank Dante D{\textquoteright}India for fish care. We thank Brian Link and Erez Raz for reagents. We thank Louise Trakimas, Elizabeth Benecchi, and Margaret Coughlin for assistance with electron microscopy at the Harvard Medical School EM facility. Lisa Goodrich, Becky Ward, Amelia Green, Akankshi Munjal, Sandra Swinburne, and the Megason lab for comments. Kerry Sobieski, Betzig lab members, and members of the Janelia Research Campus aquatics facility for helping with travel, shipping, and assisting with experiments at Janelia Research Campus. IAS was supported by NIH fellowship 5F32HL097599, a Hearing Health Foundation Emerging Research Grant, and a Novartis Fellowship in Systems Biology. KRM was supported by NIH grant K25 HD071969. DGCH was supported by NIH grants T32 MH20017 and T32 HL007901 and NSF IIA EAPSI award 1317014. TK acknowledges support from the Janelia Visitor Program, HHMI, Biogen and NIH grant R01 GM075252. SU is a Fellow at the Image and Data Analysis Core at Harvard Medical School. This work was supported by R01 DC010791 and R01 DC015478 from the National Institute of Deafness and Other Communication Disorders (SGM) and by NIH grants DP1 NS082121, RC2 NS069407 from the NINDS (FE). National Institute of Neurological Disorders and Stroke Florian Engert. Funding Information: We thank Dante D{\textquoteright}India for fish care. We thank Brian Link and Erez Raz for reagents. We thank Louise Trakimas, Elizabeth Benecchi, and Margaret Coughlin for assistance with electron microscopy at the Harvard Medical School EM facility. Lisa Goodrich, Becky Ward, Amelia Green, Akankshi Mun-jal, Sandra Swinburne, and the Megason lab for comments. Kerry Sobieski, Betzig lab members, and members of the Janelia Research Campus aquatics facility for helping with travel, shipping, and assisting with experiments at Janelia Research Campus. IAS was supported by NIH fellowship 5F32HL097599, a Hearing Health Foundation Emerging Research Grant, and a Novartis Fellowship in Systems Biology. KRM was supported by NIH grant K25 HD071969. DGCH was supported by NIH grants T32 MH20017 and T32 HL007901 and NSF IIA EAPSI award 1317014. TK acknowledges support from the Janelia Visitor Program, HHMI, Biogen and NIH grant R01 GM075252. SU is a Fellow at the Image and Data Analysis Core at Harvard Medical School. This work was supported by R01 DC010791 and R01 DC015478 from the National Institute of Deafness and Other Communication Disorders (SGM) and by NIH grants DP1 NS082121, RC2 NS069407 from the NINDS (FE). Publisher Copyright: {\textcopyright} Swinburne et al.",
year = "2018",
month = jun,
day = "19",
doi = "10.7554/eLife.37131",
language = "English",
volume = "7",
journal = "eLife",
issn = "2050-084X",
}