Analysis Tools
mortality/aging
|
• all mice die by E16.5
|
|
• fewer than expected mice are present between E11.5 and E14.5
|
nervous system
|
• absence of floor plate in the posterior spinal cord, as determined by marker expression
|
|
• slightly in the anterior spinal cord
|
|
• at E10.5, 38 of 53 mice exhibit a tight mesencephalic flexure (kink), unlike wild-type mice
|
|
• at E11.5, 20 of 53 mice exhibit an expansion of the dorsal diencephalon compared with wild-type mice
|
|
• in 20 of 53 mice at E11.5
|
exencephaly
(
J:159120
)
|
• at E10.5 in 15 of 53 mice
• however, the spinal cord and hindbrain are closed properly
|
|
• in the posterior spinal cord, the motor neuron domain is expanded ventrally to occupy the ventral midline of the spinal cord, unlike in wild-type mice
• in the anterior spinal cord, although most motor neurons remain in their normal ventral-lateral location, a few motor neurons are mislocalized in the ventral-most region
|
|
• in the posterior spinal cord, V3 interneurons are located in the ventral midline as opposed to next to the floor plate as in wild-type mice
• in the anterior spinal cord, V3 interneurons are ectopically expressed in the ventral midline compared to in wild-type mice
|
embryo
|
• although primary cilia are present on most ventral embryonic node cells at E8.0, many cilia are severely shorter than wild-type cilia
|
|
• abnormal patterning of the ventral spinal cord with loss or reduction of the floor plate accompanied by ventral expansion of V3 interneurons and motor neurons
• patterning defect is more obvious in the posterior and less severe in the anterior region of the spinal cord
|
|
• abnormal anterior-posterior patterning of the limb buds
|
|
• only a small number of cells are ciliated in E10.5 limb buds, unlike in wild-type controls
|
|
• in mice with closed neural tubes at E11.5
|
|
• absence of floor plate in the posterior spinal cord, as determined by marker expression
|
|
• slightly in the anterior spinal cord
|
|
• the notochord is closer to the neural tube than in wild-type mice
|
homeostasis/metabolism
|
• in 9 of 53 mice
|
limbs/digits/tail
|
• only a small number of cells are ciliated in E10.5 limb buds, unlike in wild-type controls
|
|
• in mice with closed neural tubes at E11.5
|
polydactyly
(
J:159120
)
|
• severe with 8 to 9 digits in the forelimb and 7 to 8 digits in the hindlimb
|
cellular
|
• a reduced number of cilia are present on the ventricular layer the spinal cord, in limb buds and other tissues at E10.5
• in culture, serum starved fibroblasts derived from E12.5 limb buds fail to develop cilia, unlike similarly treated wild-type cells
• however, cilia formation is rescued by over-expression of a cytoplasmic GFP-Intu fusion protein
|
|
• although primary cilia are present on most ventral embryonic node cells at E8.0, many cilia are severely shorter than wild-type cilia
|
vision/eye
|
• in mice with closed neural tubes at E11.5
|
cardiovascular system
|
• in 1 of 53 mice
|
|
• in 9 of 53 mice
|
