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PMID:10409504
| Citation |
Engelkamp, D, Rashbass, P, Seawright, A and van Heyningen, V (1999) Role of Pax6 in development of the cerebellar system. Development 126:3585-96 |
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| Abstract |
Post-mitotic neurons generated at the rhombic lip undertake long distance migration to widely dispersed destinations, giving rise to cerebellar granule cells and the precerebellar nuclei. Here we show that Pax6, a key regulator in CNS and eye development, is strongly expressed in rhombic lip and in cells migrating away from it. Development of some structures derived from these cells is severely affected in Pax6-null Small eye (Pax6(Sey)/Pax6(Sey)) embryos. Cell proliferation and initial differentiation seem unaffected, but cell migration and neurite extension are disrupted in mutant embryos. Three of the five precerebellar nuclei fail to form correctly. In the cerebellum the pre-migratory granule cell sub-layer and fissures are absent. Some granule cells are found in ectopic positions in the inferior colliculus which may result from the complete absence of Unc5h3 expression in Pax6(Sey)/Pax6(Sey) granule cells. Our results suggest that Pax6 plays a strong role during hindbrain migration processes and at least part of its activity is mediated through regulation of the netrin receptor Unc5h3. |
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| Keywords |
Animals; Cell Differentiation; Cell Division; Cell Movement; Cerebellum/cytology; Cerebellum/embryology; Crosses, Genetic; DNA-Binding Proteins/analysis; DNA-Binding Proteins/genetics; DNA-Binding Proteins/metabolism; Epithelial Cells/cytology; Epithelial Cells/physiology; Eye Abnormalities/genetics; Eye Proteins/genetics; Eye Proteins/metabolism; Homeodomain Proteins; Mice; Mice, Knockout; Neurites/physiology; Neurons/cytology; Neurons/physiology; Organ Culture Techniques; Paired Box Transcription Factors; Repressor Proteins; Transcription Factors/deficiency; Transcription Factors/genetics; Transcription Factors/metabolism |
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Significance
Annotations
| Gene product | Qualifier | GO ID | GO term name | Evidence Code | with/from | Aspect | Notes | Status |
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