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PMID:17395647

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Citation

Bok, J, Dolson, DK, Hill, P, Rüther, U, Epstein, DJ and Wu, DK (2007) Opposing gradients of Gli repressor and activators mediate Shh signaling along the dorsoventral axis of the inner ear. Development 134:1713-22

Abstract

Organization of the vertebrate inner ear is mainly dependent on localized signals from surrounding tissues. Previous studies demonstrated that sonic hedgehog (Shh) secreted from the floor plate and notochord is required for specification of ventral (auditory) and dorsal (vestibular) inner ear structures, yet it was not clear how this signaling activity is propagated. To elucidate the molecular mechanisms by which Shh regulates inner ear development, we examined embryos with various combinations of mutant alleles for Shh, Gli2 and Gli3. Our study shows that Gli3 repressor (R) is required for patterning dorsal inner ear structures, whereas Gli activator (A) proteins are essential for ventral inner ear structures. A proper balance of Gli3R and Gli2/3A is required along the length of the dorsoventral axis of the inner ear to mediate graded levels of Shh signaling, emanating from ventral midline tissues. Formation of the ventral-most otic region, the distal cochlear duct, requires robust Gli2/3A function. By contrast, the formation of the proximal cochlear duct and saccule, which requires less Shh signaling, is achieved by antagonizing Gli3R. The dorsal vestibular region requires the least amount of Shh signaling in order to generate the correct dose of Gli3R required for the development of this otic region. Taken together, our data suggest that reciprocal gradients of GliA and GliR mediate the responses to Shh signaling along the dorsoventral axis of the inner ear.

Links

PubMed Online version:10.1242/dev.000760

Keywords

Animals; Body Patterning; Cochlear Duct/embryology; Ear, Inner/embryology; Ear, Inner/metabolism; Gene Expression Regulation, Developmental; Hedgehog Proteins/genetics; Hedgehog Proteins/metabolism; Humans; Kruppel-Like Transcription Factors/genetics; Kruppel-Like Transcription Factors/metabolism; Mice; Mutation; Nerve Tissue Proteins/genetics; Nerve Tissue Proteins/metabolism; Receptors, Cell Surface/metabolism; Repressor Proteins/metabolism; Semicircular Canals/embryology; Signal Transduction; Vestibule, Labyrinth/embryology

Significance

Annotations

Gene product Qualifier GO Term Evidence Code with/from Aspect Extension Notes Status

MOUSE:GLI2

acts_upstream_of_or_within

GO:0090103: cochlea morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1857509

P

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0060875: lateral semicircular canal development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1856276

P

  • results_in_development_of:(EMAPA:17821)

Seeded From UniProt

complete

MOUSE:GLI2

acts_upstream_of_or_within

GO:0090103: cochlea morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1857509

P

has_participant:(EMAPA:17598)

Seeded From UniProt

complete

MOUSE:GLI2

acts_upstream_of_or_within

GO:0060831: smoothened signaling pathway involved in dorsal/ventral neural tube patterning

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:95729

P

Seeded From UniProt

complete

MOUSE:SHH

acts_upstream_of_or_within

GO:0048839: inner ear development

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:95729

P

Seeded From UniProt

complete

MOUSE:SHH

acts_upstream_of_or_within

GO:0048839: inner ear development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1934261

P

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0060873: anterior semicircular canal development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1856276

P

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0048839: inner ear development

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:98297

P

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0060875: lateral semicircular canal development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1856276

P

results_in_development_of:(EMAPA:17821)

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0060831: smoothened signaling pathway involved in dorsal/ventral neural tube patterning

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:95728

P

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0007420: brain development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1856276

P

Seeded From UniProt

complete


See also

References

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