Centrin 2 is required for mouse olfactory ciliary trafficking and development of ependymal cilia planar polarity

Update item information
Publication Type pre-print
School or College School of Medicine
Department Ophthalmology
Creator Frederick, Jeanne M.
Other Author Ying, Guoxin; Avasthi, Prachee; Irwin, Mavis; Gerstner, Cecilia D.; Lucero, Mary T.; Baehr, Wolfgang
Title Centrin 2 is required for mouse olfactory ciliary trafficking and development of ependymal cilia planar polarity
Date 2014-01-01
Description Centrins are ancient calmodulin-related Ca2-binding proteins associated with basal bodies. In lower eukaryotes, Centrin2 (CETN2) is required for basal body replication and positioning, although its function in mammals is undefined. We generated a germline CETN2 knock-out (KO) mouse presenting with syndromic ciliopathy including dysosmia and hydrocephalus. Absence of CETN2 leads to olfactory cilia loss, impaired ciliary trafficking of olfactory signaling proteins, adenylate cyclase III (ACIII), and cyclic nucleotide-gated (CNG) channel, as well as disrupted basal body apical migration in postnatal olfactory sensory neurons (OSNs). In mutant OSNs, cilia baseanchoring of intraflagellar transport components IFT88, the kinesin-II subunit KIF3A, and cytoplasmic dynein 2 appeared compromised. Although the densities of mutant ependymal and respiratory cilia were largely normal, the planar polarity of mutant ependymal cilia was disrupted, resulting in uncoordinated flow of CSF. Transgenic expression of GFP-CETN2 rescued the Cetn2-deficiency phenotype. These results indicate that mammalian basal body replication and ciliogenesis occur independently of CETN2; however, mouse CETN2 regulates protein trafficking of olfactory cilia and participates in specifying planar polarity of ependymal cilia.
Type Text
Publisher Society for Neuroscience
Volume 34
Issue 18
First Page 6377
Last Page 6388
Language eng
Bibliographic Citation Ying, G., Avasthi, P., Irwin, M., Gerstner, C. D., Frederick, J. M., Lucero, M. T., & Baehr, W. (2014). Centrin 2 is required for mouse olfactory ciliary trafficking and development of ependymal cilia planar polarity. Journal of Neuroscience, 34(18), 6377-88.
Rights Management (c) Society for Neuroscience http://www.jneurosci.org/
Format Medium application/pdf
Format Extent 5,549,524 bytes
Identifier uspace,18702
ARK ark:/87278/s66q56bg
Setname ir_uspace
Date Created 2014-05-30
Date Modified 2014-05-30
ID 712576
Reference URL https://collections.lib.utah.edu/ark:/87278/s66q56bg
Back to Search Results