Analysis Tools
hearing/vestibular/ear
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• at E12.5, the ototcyst displays severe hypoplasia of the primordia of semicircular canals and a failure to elongate the endolymphatic duct, with a varying degree of severity
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• at E13.5, the mutant cochlea shows a slight growth delay which becomes evident at E16.5
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• at E16.5, the number of cochlear turns is reduced such that proper coiling is never achieved
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• at E12.5, anterior and posterior canals are rarely recongizable in mildly affected mice
• in mild cases, the epithelium precursor of canals folds (with the lateral canal often identified), but canal plates never form properly and no fusion of the central plates occurs
• in severe cases, the entire canal region is reduced to a single vesicle by a thinner single-layered epithelium
• canal malformations are associated with altered patterns of apoptosis and proliferation at E11.5, and a severe reduction of Bmp4 expression within the developing otocyst
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• at E12.5, a hypoplastic lateral canal is often recognized in mildly affected mice
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• at E13.5, no canal-like structures are identified in severely affected mice
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• at E16.5, the sensory cells of utricular maculae are sparse and disorganized
• ectopic sensory cells are also observed
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• at E12.5, the utricle is enlarged and malformed in severely affected mice
• at E16.5, the utricle is fused with the remnants of canals into a single vesicle
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• at E12.5, the saccule is slightly abnormal only in severely affected mice
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• at E16.5, the sensory cells of saccular maculae are sparse and disorganized
• ectopic sensory cells are also observed
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• at E12.5, the endolymphatic duct forms but remains shorter and broader than normal in mildly affected mice
• in severe cases, the endolymphatic duct remains rudimentary
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