Analysis Tools
integument
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• the percentage of awl and auchene hairs is significantly reduced from 9.2% +/- 2.2% and 9.5% +/- 3.1%, respectively, in heterozygous mice to 1.8% +/- 1.9% and 2.8% +/- 1.7%, respectively, in homozygous mice
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• the percentage of awl and auchene hairs is significantly reduced from 9.2% +/- 2.2% and 9.5% +/- 3.1%, respectively, in heterozygous mice to 1.8% +/- 1.9% and 2.8% +/- 1.7%, respectively, in homozygous mice
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• guard hairs are missing in homozygous mice, while, awl, auchene, and zigzag hairs are readily identified and showed normal shaft morphology in adults and in juveniles at 3 weeks of age
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• The percentage of zigzag hairs is increased from 79% 6 3.5% in heterozygous mice to 95.4% +/- 3.5% in homozygous mice
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• At E14.5, scanning electron microscope analysis suggests the absence of primary hair follicle primordia in homozygous embryos
• no histological evidence of dermal condensation formation in homozygous embryos
• at E16.5 most hair follicles are very small and are not associated with dermal condensations
• at E18.5 in homozygous embryos, some primary hair follicles are seen and are occasionally bifurcated, indicating an additional defect in hair follicle development
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hearing/vestibular/ear
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• mice do not exhibit any vestibular dysfunction
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• with variability in the degree of disorganization along the length of the cochlea
• the ratio of Deiters' cells and outer pillar cells to inner pillar cells is decreased compared to in control mice
• the ratio of Deiters' cells and outer pillar cells to outer hair cells is slightly increased compared to in control mice
• the ratio of inner pillar cells to inner hair cells is decreased compared to in control mice
• however, exogenous FGF rescues loss of sensory cells before E14.5
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• the ratio of outer to inner hair cells is decreased compared to in control mice
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• delayed differentiation
• mice exhibit regions of undifferentiated post-mitotic sensory cells in the lateral compartment
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• almost completely absent
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• fewer rows and total numbers
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• fewer rows
• however, the total number of inner hair cells is normal
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• almost completely absent
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• slight decrease in numbers
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• auditory brainstem response threshold is greater than 40 db above controls in the 5 to 20 kHz range
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nervous system
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• the ratio of outer to inner hair cells is decreased compared to in control mice
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• delayed differentiation
• mice exhibit regions of undifferentiated post-mitotic sensory cells in the lateral compartment
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• almost completely absent
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• fewer rows and total numbers
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• fewer rows
• however, the total number of inner hair cells is normal
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