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PMID:17714700

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Citation

Sheth, R, Bastida, MF and Ros, M (2007) Hoxd and Gli3 interactions modulate digit number in the amniote limb. Dev. Biol. 310:430-41

Abstract

During limb development, Sonic hedgehog (SHH) and HOX proteins are considered among the most important factors regulating digit number and identity. SHH signaling prevents the processing of GLI3 into a short form that functions as a strong transcriptional repressor. Gli3 mutant limbs are characterized by a severe polydactyly and associated ectopic anterior expression of 5'Hoxd genes. To genetically determine the involvement of 5'Hoxd genes in the polydactyly of Gli3 mutants, we have generated a compound mutant that simultaneously removes the three most 5'-located Hoxd genes and Gli3. Remarkably, the limbs that form in the absence of all four of these genes show the most severe polydactyly so far reported in the mouse. The analysis of gene expression performed in compound mutants allows us to propose that the increase in the number of digits is mediated by the gain in function of Hoxd10 and Hoxd9. Our results also support the notion that an adequate balance between positive and negative effects of different Hoxd genes is required for pentadactyly.

Links

PubMed Online version:10.1016/j.ydbio.2007.07.023

Keywords

Animals; Extremities/embryology; Extremities/physiology; Homeodomain Proteins/genetics; Homeodomain Proteins/metabolism; Kruppel-Like Transcription Factors/genetics; Kruppel-Like Transcription Factors/metabolism; Mice; Mutation; Nerve Tissue Proteins/genetics; Nerve Tissue Proteins/metabolism; Polydactyly/genetics; Transcription Factors/genetics; Transcription Factors/metabolism

Significance

Annotations

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

MOUSE:HXD12

acts_upstream_of_or_within

GO:0042733: embryonic digit morphogenesis

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:95729
MGI:MGI:96203
MGI:MGI:96205

P

Seeded From UniProt

complete

MOUSE:HXD11

acts_upstream_of_or_within

GO:0042733: embryonic digit morphogenesis

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:95729
MGI:MGI:96204
MGI:MGI:96205

P

Seeded From UniProt

complete

MOUSE:HXD13

acts_upstream_of_or_within

GO:0042733: embryonic digit morphogenesis

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:95729
MGI:MGI:96203
MGI:MGI:96204

P

Seeded From UniProt

complete

MOUSE:GLI3

acts_upstream_of_or_within

GO:0042733: embryonic digit morphogenesis

ECO:0000316: genetic interaction evidence used in manual assertion

MGI:MGI:96203
MGI:MGI:96204
MGI:MGI:96205

P

Seeded From UniProt

complete


See also

References

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