mortality/aging
• homozygous embryos are recovered in expected Mendelian ratios at E7.5-E9.5 but fail to turn
(J:279207)
• homozygous embryos are found in expected Mendelian ratios up to and including E9.5 but not recovered at E15.5 or at birth
(J:290235)
|
embryo
• at E8.0 and E8.5, embryonic node cells lack ARL13B-positive primary cilia
|
• at E8.0 and E8.5, cells in the neural tube lack ARL13B-positive primary cilia
|
• embryos fail to turn by E9.5
(J:279207)
• E9.5 embryos fail to turn and resemble E8.5 controls in size and in embryonic orientation
(J:290235)
|
• embryos do not progress past the size or staging of control E8.5 embryos
• however, embryos complete gastrulation and show specification of the anterior/posterior and the dorsal/ventral axis
|
• embryos are developmentally delayed at E7.5-E9.5
(J:279207)
• embryos are slightly delayed at E7.5 but fail to progress past E8.5-E9.0
(J:290235)
|
• embryos are slightly smaller than controls at E7.5 and significantly smaller by E8.5
|
• E7.5 embryos show complete lack of Shh expression in the notochordal plate; E8.0-E8.5 embryos express Shh in a pattern similar to that in E7.5-E8.0 controls, respectively
• E9.5 embryos show inconsistent presence of the notochord/notochordal plate along the midline of the A/P axis
|
• E8.75 embryos lack a FOXA2-positive floor plate
|
• E9.5 embryos show complete failure of neural tube closure; neural folds fail to hinge inward and do not make contact
|
• notochord development is irregular and discontinuous; E9.5 embryos exhibit a single discontinuous stripe of Shh expression in the presumptive notochord
• at E9.5, some FOXA2-positive notochord is identified but it is inconsistent and usually mispositioned and shifted lateral to the neural tube
• E8.75 embryos show altered notochord resolution from the dorsal midline of the endoderm; when a notochord is present, laminin is atypically distributed and alterations to the basal lamina are observed
|
growth/size/body
• embryos are developmentally delayed at E7.5-E9.5
(J:279207)
• embryos are slightly delayed at E7.5 but fail to progress past E8.5-E9.0
(J:290235)
|
• embryos are slightly smaller than controls at E7.5 and significantly smaller by E8.5
|
cardiovascular system
• embryos exhibit ballooning of the pericardial sacs
|
• E9.5 embryos exhibit an enlarged pericardial sac
|
hemorrhage
(
J:290235
)
• E9.5 embryos show pooled blood in the posterior
|
nervous system
• at E8.0 and E8.5, cells in the neural tube lack ARL13B-positive primary cilia
|
• embryos exhibit increased neural tube cell death
(J:279207)
• at E9.5, nuclei of cells within the neural tube appear fragmented and pyknotic, suggesting apoptosis
(J:290235)
• E8.5-E9.5 embryos show a drastic increase of p53-positive cells in the neural tube confirming apoptosis, whereas no/rare p53-positive cells are found in control E8.5 embryos
(J:290235)
|
• E8.75 embryos lack a FOXA2-positive floor plate
|
• E9.5 embryos show complete failure of neural tube closure; neural folds fail to hinge inward and do not make contact
|
cellular
• at E8.0 and E8.5, embryonic node cells lack ARL13B-positive primary cilia
|
• at E8.0 and E8.5, cells in the neural tube lack ARL13B-positive primary cilia
|
• E8.75 embryos show a significantly higher number of PH3-positive nuclei with a distinctive halo pattern, suggesting a cell cycle stall/arrest in prometaphase
|
• embryos exhibit increased neural tube cell death
(J:279207)
• at E9.5, nuclei of cells within the neural tube appear fragmented and pyknotic, suggesting apoptosis
(J:290235)
• E8.5-E9.5 embryos show a drastic increase of p53-positive cells in the neural tube confirming apoptosis, whereas no/rare p53-positive cells are found in control E8.5 embryos
(J:290235)
|