The Role of Zic Genes in Inner Ear Development in the Mouse: Exploring Mutant Mouse Phenotypes

Simple item page
dc.contributor.authorChervenak, Andrew P.
dc.contributor.authorBank, Lisa M.
dc.contributor.authorThomsen, Nicole
dc.contributor.authorGlanville-Jones, Hannah
dc.contributor.authorSkibo, Jonathan
dc.contributor.authorMillen, Kathleen J.
dc.contributor.authorArkell, Ruth
dc.contributor.authorBarald, Kate F.
dc.date.accessioned2015-12-10T23:22:53Z
dc.date.issued2014
dc.date.updated2015-12-10T10:35:13Z
dc.description.abstractBackground: Murine Zic genes (Zic1-5) are expressed in the dorsal hindbrain and in periotic mesenchyme (POM) adjacent to the developing inner ear. Zic genes are involved in developmental signaling pathways in many organ systems, including the ear, although their exact roles haven't been fully elucidated. This report examines the role of Zic1, Zic2, and Zic4 during inner ear development in mouse mutants in which these Zic genes are affected. Results: Zic1/Zic4 double mutants don't exhibit any apparent defects in inner ear morphology. By contrast, inner ears from Zic2kd/kd and Zic2Ku/Ku mutants have severe but variable morphological defects in endolymphatic duct/sac and semicircular canal formation and in cochlear extension in the inner ear. Analysis of otocyst patterning in the Zic2Ku/Ku mutants by in situ hybridization showed changes in the expression patterns of Gbx2 and Pax2. Conclusions: The experiments provide the first genetic evidence that the Zic genes are required for morphogenesis of the inner ear. Zic2 loss-of-function doesn't prevent initial otocyst patterning but leads to molecular abnormalities concomitant with morphogenesis of the endolymphatic duct. Functional hearing deficits often accompany inner ear dysmorphologies, making Zic2 a novel candidate gene for ongoing efforts to identify the genetic basis of human hearing loss.
dc.identifier.issn1058-8388
dc.identifier.urihttp://hdl.handle.net/1885/66707
dc.publisherJohn Wiley & Sons Inc
dc.sourceDevelopmental Dynamics
dc.titleThe Role of Zic Genes in Inner Ear Development in the Mouse: Exploring Mutant Mouse Phenotypes
dc.typeJournal article
local.bibliographicCitation.lastpage1498
local.bibliographicCitation.startpage1487
local.contributor.affiliationChervenak, Andrew P., University of Michigan Medical School
local.contributor.affiliationBank, Lisa M., University of Michigan Medical School
local.contributor.affiliationThomsen, Nicole, College of Medicine, Biology and Environment, ANU
local.contributor.affiliationGlanville-Jones, Hannah, College of Medicine, Biology and Environment, ANU
local.contributor.affiliationSkibo, Jonathan, Seattle Children's Research Institute
local.contributor.affiliationMillen, Kathleen J., Seattle Children's Research Institute
local.contributor.affiliationArkell, Ruth, College of Medicine, Biology and Environment, ANU
local.contributor.affiliationBarald, Kate F., University of Michigan Medical School
local.contributor.authoremailu4974539@anu.edu.au
local.contributor.authoruidThomsen, Nicole, u4515540
local.contributor.authoruidGlanville-Jones, Hannah, u4974539
local.contributor.authoruidArkell, Ruth, u4350791
local.description.embargo2037-12-31
local.description.notesImported from ARIES
local.identifier.absfor060103 - Cell Development, Proliferation and Death
local.identifier.absfor060403 - Developmental Genetics (incl. Sex Determination)
local.identifier.absseo970106 - Expanding Knowledge in the Biological Sciences
local.identifier.ariespublicationu9511635xPUB1329
local.identifier.citationvolume243
local.identifier.doi10.1002/DVDY.24186
local.identifier.scopusID2-s2.0-84908701392
local.identifier.thomsonID000344398400011
local.identifier.uidSubmittedByu9511635
local.type.statusPublished Version

Downloads

Original bundle
Now showing 1 - 1 of 1
Thumbnail Image
Name:
01_Chervenak_The_Role_of_Zic_Genes_in_Inner_2014.pdf
Size:
1.2 MB
Format:
Adobe Portable Document Format
Download

Collections

ANU Research Publications

DSpace software copyright © 2002-2024 LYRASIS