Abnormal corneal and eyelid development in the repeated epilation mouse.
Development of the eyelids and cornea in repeated epilation (Er/Er) mice is characterized by fusion of the tarsal conjunctiva to the epithelia of the bulbar conjunctiva and cornea. We investigated the ultrastructural features of this malformation and tested for abnormal expression of filaggrin by immunofluorescence. Heterozygous (Er/+) breeding stock were mated and 13,14,15, and 19-day-old embryos were studied by light and electron microscopy. Fusion occurred in all Er/Er specimens and was associated with abnormal migration of surface ectodermal cells onto the cornea. Immunofluorescence studies with antimouse filaggrin antibody on day 13 and day 15 revealed the presence of filaggrin precursors in the fused epithelia of mutants, but not in normal corneal or conjunctival epithelia. The results suggest defective regulation of the synthesis of cellular proteins and altered cell surface properties in the Er/Er ocular epithelia.
D000015:Abnormalities, Multiple / Q000196:embryology / Q000235:genetics*; D000818:Animals; D003315:Cornea / Q000002:abnormalities* / Q000196:embryology / Q000648:ultrastructure; D005143:Eyelids / Q000002:abnormalities* / Q000196:embryology / Q000648:ultrastructure; D000091344:Filaggrin Proteins; D005455:Fluorescent Antibody Technique; D007381:Intermediate Filament Proteins / Q000032:analysis; D051379:Mice / Q000196:embryology / Q000235:genetics*; D008854:Microscopy, Electron; D008855:Microscopy, Electron, Scanning; D009154:Mutation
D000091344:Filaggrin Proteins; D007381:Intermediate Filament Proteins