|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
| N |
• cell survival and cell division at E8.75 according to mitotic marker p-HH3 immunoreactivity
|
| N |
• brain morphology at E12.5
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• at E12.5
|
|
• reduction in size across A/P and D/V axes at E12.5
|
|
• absent at E13.5 according to lack of Lhx8- and Nkx2-1-labeling of LGE region
|
|
• absent at E13.5 according to lack of Lhx8- and Nkx2-1-labeling of MGE region
|
|
• at E12.5
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• at E12.5
|
|
• reduction in size across A/P and D/V axes at E12.5
|
|
• at E12.5
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
| N |
• cell division at E8.75 according to mitotic marker p-HH3 immunoreactivity
|
|
• according to TUNEL assay at E8.75
|
|
• at E12.5
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• maxillary processes are slightly reduced at E10.5
|
|
• frontonasal prominence is slightly reduced at E10.5
• cranial neural crest (CNC) cell participation in the frontonasal prominence is markedly reduced
• however, neural tube formation is normal
|
|
• maxillary processes are slightly reduced at E10.5
|
|
• maxillary processes are slightly reduced at E10.5
|
|
• maxillary processes are slightly reduced at E10.5
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• small
|
|
• reduced cell proliferation in genital mesenchyme
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• extensive deletions are seen in the posterior midbrain
• however, no extensive deletions or alterations are seen in the basal plate of the midbrain-hindbrain region and no change in apoptosis is seen in the dorsal midbrain at E9.5
|
|
• absence of the inferior colliculus in the posterior midbrain in newborn mice
|
|
• appears disorganized
|
|
• abnormal foliation of the hemispheres
|
|
• absent in newborns and adults
|
|
• however, in adults the trochlear motoneurons are distinguishable and the oculomotor nerve is normal in both adults and E10.5 embryos
• at E10.5 the dorsal part of the trochlear nerve can not be distinguished
|
|
• impaired performance in stationary beam and rotarod assays
|
|
• adults have a wide stance
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
| N |
• mice exhibit normal early growth, size and shape of genital tubercles
|
|
• abnormal maturation of urethral epithelium
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• underdeveloped from E11.5
• deficiency in proximodistal outgrowth at E12.5
• smaller in size at E15.5 with a lack in mesenchymal patterning
|
|
• at E11.0, genital mesenchyme exhibits a reduction in cell proliferation compared with in wild-type mice
• however, apoptosis rates are normal
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• there is about a third reduction in the length of the small intestine at E18.5 compared to controls
|
|
• premature crypt-like structures occur in the small intestine of E18.5 embryos before the appearance of paneth cells
|
|
• proliferation of fibroblasts found in the proximal and distal small intestine mesenchyme is significantly reduced at E18.5
|
|
• premature crypt-like structures occur in the small intestine of E18.5 embryos before the appearance of paneth cells
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• there is about a 15% reduction in the length of the small intestine at E18.5 compared to controls
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• there is about a 15% reduction in the length of the small intestine at E18.5 compared to controls
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• dorsal cerebellum development is abnormal
|
|
• inferior colliculus is deleted in the posterior midbrain
|
|
• the vermis is present but severely malformed
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
| N |
• at E9.5, no defects are seen in the early development of the second branchial arch unlike mice homozygous for Fgfr1tm2Jrt
|
|
• no enhancement in later craniofacial phenotypes relative to mice homozygous for Fgfr1tm1Jrt that carry the Tg(Wnt1-cre)11Rth transgene
|
|
• no enhancement in later craniofacial phenotypes relative to mice homozygous for Fgfr1tm1Jrt that carry the Tg(Wnt1-cre)11Rth transgene
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
| N |
• at E9.5, no defects are seen in the early development of the second branchial arch unlike mice homozygous for Fgfr1tm2Jrt
• also, the malleus, incus, stapes, styloid process, tympanic ring, alisphenoid and squamosum are normal
|
|
• lesser horn points laterally
|
|
• in newborns
|
|
• in newborns
|
|
• lesser horn points laterally
|
|
• in newborns
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• embryos exhibit severe tooth bud defects
|
|
• development of medial nasal processes is impaired
|
|
• cell proliferation is delayed in the mesenchymal compartment of palate shelves; BrdU incorporation assays showed reduced cell proliferation at E12.5-13.5 but enhanced proliferation at E14.5
|
|
• immunostaining of coronal sections of E14.5 embryos with anti-phosphorylated Smad1/5/8 showed increased BMP signaling especially in the anterior part of palate shelves
• cell proliferation is delayed in both the epithelial and mesenchymal compartments of palate shelves; BrdU incorporation assays showed reduced cell proliferation at E12.5-13.5 but enhanced proliferation at E14.5
|
|
• at E14.5, palatal shelves are widely separated allowing direct view of the presphenoid bone
|
|
• ex vivo, dissected E13.5 palate shelves placed in proximal apposition do fuse after 2 days in culture but the gap between the two shelves is not fully filled by mesenchymal cells and a fraction of midline epithelial seam is still present while expression of epithelial cytokeratins is not completely eliminated in the medial edge epithelium (MEE), unlike in control palate shelves
• degeneration of the MEE is compromised, as shown by less defused E-cadherin staining and a lower number of TUNEL+ apoptotic cells in MEE cells at E14.5 and reduced expression of Tgfb3 at E13.5
|
|
• palate shelves appear smaller at E13.5
|
|
• complete cleft lip observed at E14.5 and P0
|
|
• complete cleft primary palate observed at E14.5 and P0
|
|
• partially formed palate rugae observed at P0
|
|
• complete secondary palate observed at E14.5 and P0
|
|
• palate shelves continue to grow downward at both anterior and posterior positions at E14.5 and fail to elevate and complete fusion even at E15.5
|
|
• tongue position is heightened at E14.5
|
|
• cell proliferation is delayed in the mesenchymal compartment of palate shelves; BrdU incorporation assays showed reduced cell proliferation at E12.5-13.5 but enhanced proliferation at E14.5
|
|
• immunostaining of coronal sections of E14.5 embryos with anti-phosphorylated Smad1/5/8 showed increased BMP signaling especially in the anterior part of palate shelves
• cell proliferation is delayed in both the epithelial and mesenchymal compartments of palate shelves; BrdU incorporation assays showed reduced cell proliferation at E12.5-13.5 but enhanced proliferation at E14.5
|
|
• at E14.5, palatal shelves are widely separated allowing direct view of the presphenoid bone
|
|
• ex vivo, dissected E13.5 palate shelves placed in proximal apposition do fuse after 2 days in culture but the gap between the two shelves is not fully filled by mesenchymal cells and a fraction of midline epithelial seam is still present while expression of epithelial cytokeratins is not completely eliminated in the medial edge epithelium (MEE), unlike in control palate shelves
• degeneration of the MEE is compromised, as shown by less defused E-cadherin staining and a lower number of TUNEL+ apoptotic cells in MEE cells at E14.5 and reduced expression of Tgfb3 at E13.5
|
|
• palate shelves appear smaller at E13.5
|
|
• complete cleft primary palate observed at E14.5 and P0
|
|
• partially formed palate rugae observed at P0
|
|
• complete secondary palate observed at E14.5 and P0
|
|
• palate shelves continue to grow downward at both anterior and posterior positions at E14.5 and fail to elevate and complete fusion even at E15.5
|
|
• tongue position is heightened at E14.5
|
|
• embryos exhibit severe tooth bud defects
|
|
• cell proliferation is delayed in the mesenchymal compartment of palate shelves; BrdU incorporation assays showed reduced cell proliferation at E12.5-13.5 but enhanced proliferation at E14.5
|
|
• immunostaining of coronal sections of E14.5 embryos with anti-phosphorylated Smad1/5/8 showed increased BMP signaling especially in the anterior part of palate shelves
• cell proliferation is delayed in both the epithelial and mesenchymal compartments of palate shelves; BrdU incorporation assays showed reduced cell proliferation at E12.5-13.5 but enhanced proliferation at E14.5
|
|
• at E14.5, palatal shelves are widely separated allowing direct view of the presphenoid bone
|
|
• ex vivo, dissected E13.5 palate shelves placed in proximal apposition do fuse after 2 days in culture but the gap between the two shelves is not fully filled by mesenchymal cells and a fraction of midline epithelial seam is still present while expression of epithelial cytokeratins is not completely eliminated in the medial edge epithelium (MEE), unlike in control palate shelves
• degeneration of the MEE is compromised, as shown by less defused E-cadherin staining and a lower number of TUNEL+ apoptotic cells in MEE cells at E14.5 and reduced expression of Tgfb3 at E13.5
|
|
• palate shelves appear smaller at E13.5
|
|
• complete cleft lip observed at E14.5 and P0
|
|
• complete cleft primary palate observed at E14.5 and P0
|
|
• partially formed palate rugae observed at P0
|
|
• complete secondary palate observed at E14.5 and P0
|
|
• palate shelves continue to grow downward at both anterior and posterior positions at E14.5 and fail to elevate and complete fusion even at E15.5
|
|
• tongue position is heightened at E14.5
|
|
• embryos exhibit severe tooth bud defects
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• severe facial clefting is seen
|
| N |
• despite ubiquitous expression of these genes in the anterior of the embryo, no defects in outflow tract development are seen
|
|
• severe facial clefting is seen
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
| N |
• mice exhibit normal-appearing kidneys after birth
• kidneys exhibit normal cortical and medullary structures at E16.5 and remain normal throughout embryonic development
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
| N |
• mice exhibit normal-appearing kidneys after birth
• kidneys exhibit normal cortical and medullary structures at E16.5 and remain normal throughout embryonic development
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• at E10.5, many apoptotic (TUNEL+) cells are present in the very loose mesenchyme adjacent to the ureteric bud, unlike in controls
|
|
• abnormally high rates of apoptosis are detected in mesenchyme dorsal to the initial ureteric bud at E10.5 and then in the rudimentary ureteric bud and Wolffian duct at E11.5
|
|
• at E10.5, mice exhibit local areas of hypoproliferation (decreased BrdU uptake) in mesenchyme dorsal to the ureteric bud (where metanephric mesenchyme should be present)
|
|
• at E10.5, mice exhibit no histologically recognizable condensing metanephric mesenchyme within the loose mesenchyme, unlike controls
• by E11.5, no definitive metanephric mesenchyme is seen around the unbranched ureteric buds, unlike in controls
|
|
• mice exhibit total renal aplasia
• however, mice develop bladders and structures originating from intermediate mesoderm including ovaries, testes, Mullerian ducts, and ductus deferens
|
|
• mice occasionally develop two initial ureteric buds from the Wolffian duct, unlike controls
|
|
• failure of uteretic buds to branch by E11.5
|
|
• by E11.5, ureteric buds fail to elongate or branch
|
|
• mice occasionally develop an ectopic ureteric bud
|
|
• a smaller ureteric bud is usually observed at E10.5 and E11.0
|
|
• at E10.5, many apoptotic (TUNEL+) cells are present in the very loose mesenchyme adjacent to the ureteric bud, unlike in controls
|
|
• abnormally high rates of apoptosis are detected in mesenchyme dorsal to the initial ureteric bud at E10.5 and then in the rudimentary ureteric bud and Wolffian duct at E11.5
|
|
• at E10.5, mice exhibit local areas of hypoproliferation (decreased BrdU uptake) in mesenchyme dorsal to the ureteric bud (where metanephric mesenchyme should be present)
|
|
• at E10.5, many apoptotic (TUNEL+) cells are present in the very loose mesenchyme adjacent to the ureteric bud, unlike in controls
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• at E10.5, mice exhibit a severe reduction in the size of condensed metanephric mesenchyme relative to controls, as shown by H&E staining
• still images of 3D reconstructions show a 70% reduction in metanephric mesenchyme volume at E10.5
• at E11.5, the metanephric mesenchyme remains small and less condensed, unlike in controls
|
|
• by E12.5, high levels of apoptosis are noted throughout the remnant metanephric mesenchyme, as revealed by TUNEL staining
• however, no obvious differences in mesenchymal proliferation or apoptosis are noted at E10.5 and E11.5 relative to controls
|
|
• no apparent kidney tissues are observed at E13.5 and E18.5
|
|
• at E11.5, ureteric buds remain unbranched, unlike in controls
|
|
• at E11.5, none of the ureteric buds elongate into the metanephric mesenchyme, unlike in controls
|
|
• a subset of mice develop an anteriorly displaced secondary ureteric bud, unlike in controls
|
|
• at E11.5, ureteric buds are small and unbranched, unlike in controls
|
|
• by E12.5, high levels of apoptosis are noted throughout the remnant metanephric mesenchyme, as revealed by TUNEL staining
• however, no obvious differences in mesenchymal proliferation or apoptosis are noted at E10.5 and E11.5 relative to controls
|
|
• by E12.5, high levels of apoptosis are noted throughout the remnant metanephric mesenchyme, as revealed by TUNEL staining
• however, no obvious differences in mesenchymal proliferation or apoptosis are noted at E10.5 and E11.5 relative to controls
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• reduced numbers of glomeruli in both embryonic and adult kidneys
|
|
• some adult mutants had hydronephrosis occurring unilaterally or bilaterally
|
|
• E13.5 and E16.5 kidneys were smaller than controls
• many adult mutants had small, abnormally shaped kidneys
|
|
• E11.5 ureteric bud explants had abnormally thin, long ureteric stalks and fewer peripheral tips, with a range in phenotype severity
• decrease in the mean number of ureteric bud tips on the surface of E16.5 kidneys
• range of ureteric bud defects at E16.5, including extremely small kidneys with large areas devoid of ureteric tissue
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
| N |
• normal kidneys and no apparent renal abnormalities
|
|
|
| ♀ | phenotype observed in females |
| ♂ | phenotype observed in males |
| N | normal phenotype |
|
• 22% and 24% fewer glomeruli in E11.5 explants and adult kidneys, respectively
|
|
• defects in cortical stromal mesenchyme patterning, showing aberrantly thickened stroma in subcapsular regions in E13.5 kidneys and regions of massive subcapsular apoptosis in stroma
• absent intercalated stripes of stromal cells in E13.5 kidneys
|
|
• 20% of adult mutants had hydronephrosis occurring unilaterally or bilaterally
|
|
• E13.5 and E16.5 kidneys were smaller than controls
• 80% of adult mutants had small, abnormally shaped kidneys
|
|
• range of ureteric bud defects at E16.5, including extremely small kidneys with large areas devoid of ureteric tissue
• increased apoptotic nuclei in ureteric buds (2-3 apoptotic nuclei compared to none or just one in controls)
• decreased rates of proliferation in ureteric bud tips compared with controls
|
|
• E11.5 ureteric bud explants exhibited aberrant branching and had abnormally thin, long ureteric stalks and fewer peripheral tips, with a range in phenotype severity
• decrease in the mean number of ureteric bud tips on the surface of E16.5 kidneys (89.5 versus 271 in control)
|
Mouse Genome Database (MGD), Gene Expression Database (GXD), Mouse Models of Human Cancer database (MMHCdb) (formerly Mouse Tumor Biology (MTB)), Gene Ontology (GO) |
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last database update 04/30/2024 MGI 6.23 |
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