Analysis Tools
mortality/aging
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• mice occasionally survive to E16.5
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• most mice die at midgestation from E10.5 to E12.5
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vision/eye
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• at E12.5
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growth/size/body
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• delayed embryonic development at E10.5
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embryo
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• delayed embryonic development at E10.5
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• at E10.5, expression of limb-patterning genes is altered in the limb buds; expression of the direct Hh targets, Gli1 and Ptch1, is not detected, while Shh is normally expressed
• expression of limb-patterning genes that are normally restricted to the posterior region by the Gli3 repressor (Gremlin, Hand2, Hoxd11, Hoxd13) is anteriorly expanded
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• at E10.5, expression of both Fgf4 and Fgf8 in the apical ectodermal ridge (AER), normally restricted by the Gli3 repressor, is anteriorly extended
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• at E10.5, cilia in limb mesenchymal cells are short and stumpy
• however, cilia density in the limb mesenchyme is similar to that in wild-type controls
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• at E10.5, Shh-dependent neural patterning in the neural tube is disrupted while Hh signaling is inhibited, unlike in wild-type embryos
• ventral neural progenitors are not specified at E10.5; none of the ventral markers (Foxa2, Nkx2.2, Hb9, Isl1/2) are detected while expression of dorsal markers (Pax6 and Pax7) is ventrally expanded
• however, Shh expression in the notochord is normal
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• at E9.5-E10.5, cilia of neuroepithelia in the neural tube are short and swollen (stumpy)
• however, cilia density in neuroepithelia is similar to that in wild-type controls
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spina bifida
(
J:242833
)
nervous system
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• at E10.5, Shh-dependent neural patterning in the neural tube is disrupted while Hh signaling is inhibited, unlike in wild-type embryos
• ventral neural progenitors are not specified at E10.5; none of the ventral markers (Foxa2, Nkx2.2, Hb9, Isl1/2) are detected while expression of dorsal markers (Pax6 and Pax7) is ventrally expanded
• however, Shh expression in the notochord is normal
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• at E9.5-E10.5, cilia of neuroepithelia in the neural tube are short and swollen (stumpy)
• however, cilia density in neuroepithelia is similar to that in wild-type controls
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spina bifida
(
J:242833
)
exencephaly
(
J:242833
)
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• open brain at E12.5
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limbs/digits/tail
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• at E10.5, expression of limb-patterning genes is altered in the limb buds; expression of the direct Hh targets, Gli1 and Ptch1, is not detected, while Shh is normally expressed
• expression of limb-patterning genes that are normally restricted to the posterior region by the Gli3 repressor (Gremlin, Hand2, Hoxd11, Hoxd13) is anteriorly expanded
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• at E10.5, expression of both Fgf4 and Fgf8 in the apical ectodermal ridge (AER), normally restricted by the Gli3 repressor, is anteriorly extended
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• at E10.5, cilia in limb mesenchymal cells are short and stumpy
• however, cilia density in the limb mesenchyme is similar to that in wild-type controls
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polydactyly
(
J:242833
)
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• at E12.5 and E16.5
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• wider limb paddle at E12.5, indicating polydactyly
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cellular
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• cilia of mouse embryo fibroblasts (MEFs) derived from E11.5 or E12.5 embryos are short, stumpy and significantly reduced in number relative to cilia of wild-type MEFs
• in MEFs, all ciliary proteins examined (Arl13b, Smo, Ift88, Ift57, and Ift140) accumulate abnormally at the distal cilia tip, indicating disrupted retrograde ciliary protein trafficking
• however, centriolar localization of centrosomal proteins is normal
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• at E9.5-E10.5, cilia of neuroepithelia in the neural tube are short and swollen (stumpy)
• however, cilia density in neuroepithelia is similar to that in wild-type controls
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• nocodazole-treated MEFs show microtubule regrowth delays after nocodazole removal relative to similarly treated wild-type MEFs
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