mortality/aging
• fewer homozygous embryos than expected at E15.5 to E18.5
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• most of mutant mice surviving to birth die a few hours after birth with respiratory defects
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nervous system
• low concentration (0.1 mM) sphingosine-1-phosphate (S1P) and lysophosphatidic acid induce contraction of mutant neural progenitor cells isolated from telencephalons at E15.5 to E18.5
• absent actin-rich protrusions in S1P stimulated mutant neural progenitor cells staining with phalloidin
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• reduced endothelin-1-induced and sphingosine-1-phosphate-induced proliferation of neural progenitor cells isolated from mutant telencephalons at E15.5 to E18.5
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• abnormal dorsolateral bending of cranial neural plate at the stage just before neural tube closure (E8.75)
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• elongated neuroepithelial cells and nuclei along the apical-basal axis at E8.75
• reduced F-actin staining in the basal side neuroepithelium around the dorsolateral hinge points in the mutant at E9.5
• normal F-actin staining in the apical side
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• severe brain malformations in mutant embryos with neural tube defects
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• reduced number of pH3-positive cells in the ventriclar zone of the cerebral cortex in mutant embryos at E16.5 but not at E10.5
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• smaller brain volumes at E18.5
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• reduced cortical thickness at P0
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exencephaly
(
J:157907
)
• midbrain-hindbrain exencephaly in about 40% of mutant embryos at E10.5
• no spina bifida
• exencephaly in about 30% of mutant embryos at E18.5
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respiratory system
• most of mutant mice surviving to birth die a few hours after birth with respiratory defects
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behavior/neurological
• some of the mutant die within 2 days after birth since they could not ingest milk
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embryo
• abnormal dorsolateral bending of cranial neural plate at the stage just before neural tube closure (E8.75)
|
• elongated neuroepithelial cells and nuclei along the apical-basal axis at E8.75
• reduced F-actin staining in the basal side neuroepithelium around the dorsolateral hinge points in the mutant at E9.5
• normal F-actin staining in the apical side
|
cellular
• low concentration (0.1 mM) sphingosine-1-phosphate (S1P) and lysophosphatidic acid induce contraction of mutant neural progenitor cells isolated from telencephalons at E15.5 to E18.5
• absent actin-rich protrusions in S1P stimulated mutant neural progenitor cells staining with phalloidin
|
• reduced endothelin-1-induced and sphingosine-1-phosphate-induced proliferation of neural progenitor cells isolated from mutant telencephalons at E15.5 to E18.5
|
• reduced number of pH3-positive cells in the ventriclar zone of the cerebral cortex in mutant embryos at E16.5 but not at E10.5
|