Cell Surface Glycoconjugate Abnormalities and Corneal Epithelial Wound Healing in thePax6+/−Mouse Model of Aniridia-Related Keratopathy
Open Access
- 1 December 2006
- journal article
- cornea
- Published by Association for Research in Vision and Ophthalmology (ARVO)
- Vol. 47 (12), 5276-5282
- https://doi.org/10.1167/iovs.06-0581
Abstract
Purpose. Congenital aniridia due to heterozygosity for Pax6 is associated with ocular surface disease, including keratopathy. This study investigated how defects in glycoconjugate component of the cell surface of Pax6 +/− could cause the abnormal cellular migration phenotypes associated with the disease. methods. Immunohistochemistry, lectin-based histochemistry, conventional staining techniques, and proteomic assays were performed on eyes and cultured corneal epithelial cells from wild-type and Pax6 +/ − littermates. Wild-type cells were manipulated in culture to replicate the glycoconjugate abnormalities found in Pax6 heterozygotes and determine the consequences for wound healing. results. Multiple glycoconjugate defects were found in Pax6-mutant cells. Lectin cytochemistry of corneal epithelial cells suggested a partial failure of glycoprotein trafficking. Blocking cell surface carbohydrate moieties in wild-type corneal cells caused wound-healing delays similar to those seen in untreated Pax6 +/ − cells. conclusions. Alterations to the cell surface glycoconjugate signature of Pax6 +/ − corneal epithelia restrict the ability of cells to initiate migration in response to wounding. This underlies the observed wound-healing delay in cultured Pax6 +/ − epithelia.Keywords
This publication has 37 references indexed in Scilit:
- Increased Apoptosis and Abnormal Wound-Healing Responses in the HeterozygousPax6+/−Mouse CorneaPublished by Association for Research in Vision and Ophthalmology (ARVO) ,2006
- Accelerated Wound Healing of Alkali-Burned Corneas in MRL Mice Is Associated with a Reduced Inflammatory SignaturePublished by Association for Research in Vision and Ophthalmology (ARVO) ,2005
- Nerve regeneration and wound healing are stimulated and directed by an endogenous electrical field in vivoJournal of Cell Science, 2004
- Transcription Factors Pax6 and AP-2α Interact To Coordinate Corneal Epithelial Repair by Controlling Expression of Matrix Metalloproteinase Gelatinase BMolecular and Cellular Biology, 2004
- Requirement for Pax6 in corneal morphogenesis: a role in adhesionJournal of Cell Science, 2003
- Corneal Abnormalities inPax6+/−Small Eye Mice Mimic Human Aniridia-Related KeratopathyPublished by Association for Research in Vision and Ophthalmology (ARVO) ,2003
- Glycosaminoglycans in the study of mammalian organ developmentBiochemical Society Transactions, 2001
- Ultrastructurallmmunohistological and Biochemical Characterization of Cultured Mouse Corneal Epithelial CellsOphthalmic Research, 1996
- Colocalization of thrombospondin and syndecan during murine developmentDevelopmental Dynamics, 1992
- Mouse Small eye results from mutations in a paired-like homeobox-containing geneNature, 1991