Foxg1 is required for morphogenesis and histogenesis of the mammalian inner ear

Sarah Pauley; Eseng Lai; Bernd Fritzsch

doi:10.1002/dvdy.20839

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Foxg1 is required for morphogenesis and histogenesis of the mammalian inner ear

Journal article

Open access

Peer reviewed

Foxg1 is required for morphogenesis and histogenesis of the mammalian inner ear

Sarah Pauley, Eseng Lai and Bernd Fritzsch

Developmental dynamics, Vol.235(9), pp.2470-2482

09/2006

DOI: 10.1002/dvdy.20839

PMCID: PMC3901532

PMID: 16691564

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Abstract

The forkhead genes are involved in patterning, morphogenesis, cell fate determination, and proliferation. Several Fox genes (Foxi1, Foxg1) are expressed in the developing otocyst of both zebrafish and mammals. We show that Foxg1 is expressed in most cell types of the inner ear of the adult mouse and that Foxg1 mutants have both morphological and histological defects in the inner ear. These mice have a shortened cochlea with multiple rows of hair cells and supporting cells. Additionally, they demonstrate striking abnormalities in cochlear and vestibular innervation, including loss of all crista neurons and numerous fibers that overshoot the organ of Corti. Closer examination shows that some anterior crista fibers exist in late embryos. Tracing these fibers shows that they do not project to the brain but, instead, to the cochlea. Finally, these mice completely lack a horizontal crista, although a horizontal canal forms but comes off the anterior ampulla. Anterior and posterior cristae, ampullae, and canals are reduced to varying degrees, particularly in combination with Fgf10 heterozygosity. Compounding Fgf10 heterozygotic effects suggest an additive effect of Fgf10 on Foxg1, possibly mediated through bone morphogenetic protein regulation. We show that sensory epithelia formation and canal development are linked in the anterior and posterior canal systems. Much of the Foxg1 phenotype can be explained by the participation of the protein binding domain in the delta/notch/hes signaling pathway. Additional Foxg1 effects may be mediated by the forkhead DNA binding domain.

Epithelial Cells - metabolism

Ear, Inner - embryology

Nerve Tissue Proteins - deficiency

Forkhead Transcription Factors - physiology

Tubulin - metabolism

Gene Expression Regulation, Developmental

Female

Forkhead Transcription Factors - deficiency

Hair Cells, Auditory - abnormalities

Nerve Tissue Proteins - physiology

Morphogenesis - genetics

Ear, Inner - cytology

Mice, Transgenic

Forkhead Transcription Factors - genetics

Nerve Tissue Proteins - genetics

Mice, Knockout

Pregnancy

Ear, Inner - metabolism

Phenotype

Animals

Ear, Inner - abnormalities

Heterozygote

Mice

Cochlea - abnormalities

Neurons, Afferent - metabolism

Morphogenesis - physiology

Details

Title: Subtitle: Foxg1 is required for morphogenesis and histogenesis of the mammalian inner ear
Creators: Sarah Pauley - Creighton University, Department of Biomedical Sciences, Omaha, Nebraska 68178, USA
Eseng Lai
Bernd Fritzsch
Resource Type: Journal article
Publication Details: Developmental dynamics, Vol.235(9), pp.2470-2482
DOI: 10.1002/dvdy.20839
PMID: 16691564
PMCID: PMC3901532
NLM abbreviation: Dev Dyn
ISSN: 1058-8388
eISSN: 1097-0177
Publisher: United States
Grant note: 1P20RR018788-01 / NCRR NIH HHS P20 RR018788 / NCRR NIH HHS 1 C06 RR17417-01 / NCRR NIH HHS R01 DC005590 / NIDCD NIH HHS C06 RR017417 / NCRR NIH HHS R01 DC005590-05 / NIDCD NIH HHS
Language: English
Date published: 09/2006
Academic Unit: Iowa Neuroscience Institute; Biology; Craniofacial Anomalies Research Center
Record Identifier: 9984070483802771

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