nervous system
• at E13.5
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vision/eye
• at E13.5
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Allele Symbol Allele Name Allele ID |
Atoh7tm2Gan targeted mutation 2, Lin Gan MGI:2159014 |
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Summary |
6 genotypes
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♀ | phenotype observed in females |
♂ | phenotype observed in males |
N | normal phenotype |
• at E13.5
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• at E13.5
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♀ | phenotype observed in females |
♂ | phenotype observed in males |
N | normal phenotype |
N |
• mice are fertile
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• very thin
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• very thin
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• the overall structure of retina and number of photoreceptor cells is normal
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• retinal ganglion cells fail to differentiate as shown by the loss of Pou4f1+ and reduction of Ngfr+ cells
• approximately 97% of Pou4f2+ cells are lost
• 80% reduction in the SMI-32 positive processes of axons from large ganglion cells and the remaining 20% of SMI-32 positive processes of axons have abnormally curved axon bundles
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• greater than 80% reduction in the number of retinal ganglion cells
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• absence of a defined nerve fiber layer
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• retinas are approximately 15 to 20% thinner than control retinas
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• retinal ganglion cells fail to differentiate as shown by the loss of Pou4f1+ and reduction of Ngfr+ cells
• approximately 97% of Pou4f2+ cells are lost
• 80% reduction in the SMI-32 positive processes of axons from large ganglion cells and the remaining 20% of SMI-32 positive processes of axons have abnormally curved axon bundles
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• greater than 80% reduction in the number of retinal ganglion cells
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• very thin
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• very thin
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♀ | phenotype observed in females |
♂ | phenotype observed in males |
N | normal phenotype |
• hardly show either the positive or negative scotopic response waves in a dark adapted electroretinogram in response to weak stimulus
• positive scotopic threshold response amplitudes are significantly lower than in controls
• negative scotopic threshold responses are significantly lower than in controls
• b-wave amplitudes are lower than in controls
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♀ | phenotype observed in females |
♂ | phenotype observed in males |
N | normal phenotype |
• at E15.5, the ganglion cell layer contains very few differentiated ganglion cells
• however, at E11 and E15.5 the number of progenitor retinal ganglion cells are similar to that in heterozygotes and there is no evidence of increased apoptosis at E12.5 to E19
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• absence of nerve fiber layer
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• at E15.5, the ganglion cell layer contains very few differentiated ganglion cells
• however, at E11 and E15.5 the number of progenitor retinal ganglion cells are similar to that in heterozygotes and there is no evidence of increased apoptosis at E12.5 to E19
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♀ | phenotype observed in females |
♂ | phenotype observed in males |
N | normal phenotype |
• mis-patterning of the retina is more severe than in mutant mice wild-type for Atoh7
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• few ganglion cells are detected in the nascent retinal ganglion layer at E13.5 despite many being generated in the neuroblast layer suggesting a defect in migration and maturation
• many dying cells are present in the retinal ganglion cell layer at E13.5
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♀ | phenotype observed in females |
♂ | phenotype observed in males |
N | normal phenotype |
• retinas have 28% of the Isl1-positive RGCs compared to wild-type mice at E13 indicating a partial restoration of the Atoh7 null phenotype
• retinas have about 25% of the number of retinal ganglion cells as in wild-type mice
• however, cells are generated evenly across the dorsal-ventral and nasal-temporal axes
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• diameters are 20 to 40% that of wild-type optic nerves
• retinal ganglion cell axons form, fasciculate, and extend into the optic disk indicating a partial rescue of the Atoh7 null phenotype
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• dark adapted electroretinogram responses to the weakest stimuli are similar in waveform but smaller than in controls
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• retinal ganglion cell axons project to slightly more anterior areas in the superior colliculus
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• retinas have 28% of the Isl1-positive RGCs compared to wild-type mice at E13 indicating a partial restoration of the Atoh7 null phenotype
• retinas have about 25% of the number of retinal ganglion cells as in wild-type mice
• however, cells are generated evenly across the dorsal-ventral and nasal-temporal axes
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• diameters are 20 to 40% that of wild-type optic nerves
• retinal ganglion cell axons form, fasciculate, and extend into the optic disk indicating a partial rescue of the Atoh7 null phenotype
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Mouse Genome Database (MGD), Gene Expression Database (GXD), Mouse Models of Human Cancer database (MMHCdb) (formerly Mouse Tumor Biology (MTB)), Gene Ontology (GO) |
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last database update 03/25/2025 MGI 6.24 |
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