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Abnormal corneal and eyelid development in the repeated epilation mouse
Laboratory Animal Science
|April 1, 1984
Summary
Ocular development in repeated epilation (Er/Er) mice shows eyelid and cornea fusion due to abnormal cell migration. Filaggrin precursors were found in mutant epithelia, suggesting protein synthesis defects in this genetic eye malformation.
Area of Science:
- Ophthalmology
- Developmental Biology
- Genetics
Background:
- The repeated epilation (Er/Er) mouse model exhibits congenital eyelid and cornea malformations.
- Understanding the cellular and molecular basis of these ocular defects is crucial for regenerative medicine and ophthalmology.
Purpose of the Study:
- To investigate the ultrastructural characteristics of ocular malformations in Er/Er mice.
- To examine the expression of filaggrin, a key protein in epithelial differentiation, in the developing ocular tissues of Er/Er mutants.
Main Methods:
- Light and electron microscopy were used to examine ocular tissues from 13, 14, 15, and 19-day-old Er/Er mouse embryos.
- Immunofluorescence staining with an anti-mouse filaggrin antibody was performed on embryonic day 13 and 15 ocular sections.
Main Results:
- Fusion of the tarsal conjunctiva to the bulbar conjunctiva and cornea epithelia was observed in all Er/Er specimens.
- Abnormal migration of surface ectodermal cells onto the cornea was associated with the observed fusion.
- Filaggrin precursors were detected in the fused epithelia of Er/Er mutants but absent in normal corneal and conjunctival epithelia.
Conclusions:
- The Er/Er mouse model presents a distinct ocular malformation characterized by conjunctival-corneal fusion and abnormal cell migration.
- The presence of filaggrin precursors in mutant epithelia suggests a defect in the regulation of cellular protein synthesis and altered cell surface properties during ocular development.