mortality/aging
• no homozygous mutant mice are born from heterozygous intercrosses; resorptions are consistently observed at E12.5 and all stages thereafter
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embryo
• nearly complete absence of primary cilia in the neural tube at E9.5
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• truncated posterior trunk at E9.5 and E10.5
• however, embryo morphology is normal at E7.5
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cardiovascular system
• cardiac looping defects at E9.5 and E10.5
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• edematous pericardial sac at E9.5 and E10.5
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nervous system
• nearly complete absence of primary cilia in the neural tube at E9.5
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• holoprosencephaly at E9.5 and E10.5
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cellular
• dramatic loss of primary cilia in mouse embryonic fibroblasts (MEFs) derived from E8.5 embryos and serum-starved for 48 hours
• loss of CEP164, Cby1, and FAM92A centriolar localization in serum-starved MEFs
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• nearly complete absence of primary cilia in the neural tube at E9.5
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homeostasis/metabolism
• edematous pericardial sac at E9.5 and E10.5
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