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PMID:18816854

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Citation

Huang, X, Goudy, SL, Ketova, T, Litingtung, Y and Chiang, C (2008) Gli3-deficient mice exhibit cleft palate associated with abnormal tongue development. Dev. Dyn. 237:3079-87

Abstract

Palatogenesis depends on appropriate growth, elevation, and fusion of the palatal shelves and aberration in these processes can lead to palatal clefting. We observed a high incidence of palate clefting in mice deficient in Gli3, known for its role as a repressor in the absence of Shh signaling. In contrast with several current mouse models of cleft palate, Meckel's cartilage extension, cranial neural crest migration, palatal shelf proliferation, apoptosis, and key signaling components mediated by Shh, Bmp, Fgf, and Tgfbeta, appeared unaffected in Gli3-/- mice. Palatal clefting in Gli3-/- mice was consistently associated with tongue abnormalities such as failure to flatten and improper positioning, implicating a critical role of Gli3 and normal tongue morphogenesis for timely palatal shelf elevation and joining. Furthermore, Gli3-/- palatal shelves grown in roller cultures without tongue can fuse suggesting that the abnormal tongue is likely an impediment for palatal shelf joining in Gli3-/- mutants.

Links

PubMed Online version:10.1002/dvdy.21714

Keywords

Animals; Cell Proliferation; Cleft Palate/embryology; Cleft Palate/metabolism; Gene Expression Regulation, Developmental; Kruppel-Like Transcription Factors/deficiency; Kruppel-Like Transcription Factors/genetics; Kruppel-Like Transcription Factors/metabolism; Mice; Mice, Inbred C57BL; Mice, Knockout; Mutation/genetics; Nerve Tissue Proteins/deficiency; Nerve Tissue Proteins/genetics; Nerve Tissue Proteins/metabolism; Signal Transduction; Tongue/abnormalities; Tongue/cytology; Tongue/metabolism

Significance

Annotations

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

MOUSE:GLI3

acts_upstream_of_or_within

GO:0060021: roof of mouth development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1856276

P

  • results_in_development_of:(EMAPA:17930)

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0043586: tongue development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1856276

P

  • results_in_development_of:(EMAPA:17185)

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0043586: tongue development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1856276

P

results_in_development_of:(EMAPA:17185)

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0060021: roof of mouth development

ECO:0000315: mutant phenotype evidence used in manual assertion

MGI:MGI:1856276

P

results_in_development_of:(EMAPA:17930)

Seeded From UniProt

complete


See also

References

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