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.
journal_name
Dev Bioljournal_title
Developmental biologyauthors
Sheth R,Bastida MF,Ros Mdoi
10.1016/j.ydbio.2007.07.023subject
Has Abstractpub_date
2007-10-15 00:00:00pages
430-41issue
2eissn
0012-1606issn
1095-564Xpii
S0012-1606(07)01220-1journal_volume
310pub_type
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