Analysis Tools
mortality/aging
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• homozygotes die shortly after birth
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integument
| N |
• at E14.5 and E15.5, homozygotes show no apparent defects in skin development relative to wild-type controls
• no differences in epidermal stratification, differentiation, proliferation or apoptosis are noted throughout early (E14.5) and late (E18.5) stages of epidermal development
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• at E18.5, double immunofluorescence labeling of mutant hair follicles with KRT75 and AE13 revealed abnormal follicular keratinocyte differentiation
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• at E18.5, the number of proliferating (BrdU+) follicular keratinocytes in stage 2 hair follicles is markedly reduced relative to wild-type controls
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• although the number of mutant hair follicles (irrespective of their differentiation stages) is only slightly reduced at E18.5, the number of proliferating (BrdU+) follicular keratinocytes in stage 2 hair follicles is markedly reduced
• delayed hair follicle development is due to abnormal follicular keratinocyte differentiation, as shown by reduced expression of the hair follicle differentiation markers KRT75 (companion cell layer) and AE13 (hair cortex) at E18.5
• however, induction of hair follicle formation is normal, as determined by the number and expression pattern of KRT17+ hair germs at E15.5
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• at E18.5, up to 90.1% of mutant hair follicles remain in stage 1-2, whereas only 0.7% develop beyond stage 5
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• at 5 weeks post-transplantation, skin grafts of E18.5 mutant dorsal skin transplanted onto nude mice show a drastic reduction in the number of hairs due to the absence of hair follicles
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• at E18.5, the majority of mutant hair follicles are substantially reduced in size
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cellular
| N |
• immunofluorescence labeling of mutant skin with a number of ciliary markers showed normal patterns of primary cilia in hair follicles at E15.5 and E18.5
• SEM analysis of primary keratinocytes isolated from mutant embryos revealed normal formation of the ciliary axoneme
• after treatment with cytochalasin D (a compound capable of elongating the ciliary axoneme), mutant primary keratinocytes show no significant differences in the % of ciliated cells or cilium length relative to wild-type controls
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• at E18.5, double immunofluorescence labeling of mutant hair follicles with KRT75 and AE13 revealed abnormal follicular keratinocyte differentiation
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• at E18.5, the number of proliferating (BrdU+) follicular keratinocytes in stage 2 hair follicles is markedly reduced relative to wild-type controls
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