Analysis Tools
mortality/aging
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• no live homozygotes are observed at P0; embryos are recovered at a reduced Mendelian frequency at E12.5 (15.4%) and E14.5 (12.5%), suggesting mid-gestation lethality
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growth/size/body
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• embryos are developmentally delayed at E7.5
• at E6.5, expression of Foxa2, a marker of both anterior visceral endoderm (AVE) and anterior primitive streak (APS), is restricted to the distal visceral endoderm (VE) in a pattern typically seen 1 day earlier in wild-type embryos, with little enrichment in the AVE, suggesting a delay even prior to gastrulation
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• embryos are significantly smaller at E7.5 and E12.5
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• brain malformations affect primarily the anteriormost structures of the head, including the eyes
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embryo
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• embryos exhibit impaired AME specification and A/P patterning of the neural plate
• at E7.5, Foxa2 and Chordin expression remains high distally in the region of the anterior primitive streak (APS) but does not extend anteriorly in the midline axial mesendoderm, and Shh expression is weak in the anterior midline
• however, initial specification of the primary anterior-posterior axis by the AVE is normal
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• embryos exhibit anterior truncations as shown by reduced forebrain expression of Six3 and Otx2 at E8.5
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• embryos are developmentally delayed at E7.5
• at E6.5, expression of Foxa2, a marker of both anterior visceral endoderm (AVE) and anterior primitive streak (APS), is restricted to the distal visceral endoderm (VE) in a pattern typically seen 1 day earlier in wild-type embryos, with little enrichment in the AVE, suggesting a delay even prior to gastrulation
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• embryos are significantly smaller at E7.5 and E12.5
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• at E12.5, embryos show anomalies in the epithalamic and dorsal thalamic neuropeithelium
• signals produced by AME cells that orchestrate the continued patterning and morphogenesis of the anterior neuroectoderm are lost, resulting in anterior truncations
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• at E7.5, embryos exhibit conspicuous ruffling of the extraembryonic visceral yolk sac endoderm
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• at E7.5, embryos exhibit conspicuous ruffling of the extraembryonic visceral yolk sac endoderm
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nervous system
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• at E12.5, embryos show anomalies in the epithalamic and dorsal thalamic neuropeithelium
• signals produced by AME cells that orchestrate the continued patterning and morphogenesis of the anterior neuroectoderm are lost, resulting in anterior truncations
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• at E12.5, embryos lack the complex organization of the anterior forebrain
• expression of Six3 and Otx2 is reduced in the anterior forebrain at E8.5
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• at E12.5, embryos show an abnormal separation of the cerebral hemispheres, reminiscent of holoprosencephaly
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• at E12.5, the lateral ganglionic eminences appear underdeveloped
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• at E12.5, the medial ganglionic eminences appear underdeveloped
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vision/eye
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• embryos show eye defects at E12.5
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