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Canal cristae growth and fiber extension to the outer hair cells of the mouse ear require Prox1 activity
Journal article   Open access   Peer reviewed

Canal cristae growth and fiber extension to the outer hair cells of the mouse ear require Prox1 activity

Bernd Fritzsch, Miriam Dillard, Alfonso Lavado, Natasha L Harvey and Israt Jahan
PloS one, Vol.5(2), pp.e9377-e9377
02/23/2010
DOI: 10.1371/journal.pone.0009377
PMCID: PMC2826422
PMID: 20186345
url
https://doi.org/10.1371/journal.pone.0009377View
Published (Version of record) Open Access

Abstract

The homeobox gene Prox1 is required for lens, retina, pancreas, liver, and lymphatic vasculature development and is expressed in inner ear supporting cells and neurons. We have investigated the role of Prox1 in the developing mouse ear taking advantage of available standard and conditional Prox1 mutant mouse strains using Tg(Pax2-Cre) and Tg(Nes-Cre). A severe reduction in the size of the canal cristae but not of other vestibular organs or the cochlea was identified in the E18.5 Prox1(Flox/Flox); Tg(Pax2-Cre) mutant ear. In these mutant embryos, hair cell differentiated; however, their distribution pattern was slightly disorganized in the cochlea where the growth of type II nerve fibers to outer hair cells along Prox1 expressing supporting cells was severely disrupted. In the case of Nestin-Cre, we found that newborn Prox1(Flox/Flox); Tg(Nestin-Cre) exhibit only a disorganized innervation of outer hair cells despite apparently normal cellular differentiation of the organ of Corti, suggesting a cell-autonomous function of Prox1 in neurons. These results identify a dual role of Prox1 during inner ear development; growth of the canal cristae and fiber guidance of Type II fibers along supporting cells in the cochlea.
Immunohistochemistry Cochlea - embryology Ear, Inner - embryology Homeodomain Proteins - metabolism Male Vestibule, Labyrinth - metabolism Embryo, Mammalian - metabolism Epithelium - embryology In Situ Hybridization Time Factors Cochlea - metabolism Gene Expression Regulation, Developmental Tumor Suppressor Proteins - genetics Ear, Inner - ultrastructure Female Vestibule, Labyrinth - embryology Cell Differentiation Animals, Newborn Microscopy, Electron, Transmission Tumor Suppressor Proteins - metabolism Epithelium - metabolism Mice, Transgenic Homeodomain Proteins - genetics Mice, Knockout Pregnancy Microscopy, Confocal Ear, Inner - metabolism Animals Hair Cells, Auditory, Outer - metabolism Embryo, Mammalian - embryology Mice Mutation

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