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Stage-related central corneal epithelial transformation in congenital aniridia-associated keratopathy
Linköping University, Department of Clinical and Experimental Medicine, Division of Neuro and Inflammation Science. Linköping University, Faculty of Medicine and Health Sciences. Region Östergötland, Anaesthetics, Operations and Specialty Surgery Center, Department of Ophthalmology in Linköping.ORCID iD: 0000-0003-1079-4361
Medical University of Silesia, Poland.
Medical University of Silesia, Poland.
University of Oslo, Norway.
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2018 (English)In: OCULAR SURFACE, ISSN 1542-0124, Vol. 16, no 1, p. 163-172Article in journal (Refereed) Published
Abstract [en]

Purpose: To relate central corneal epithelial phenotype to degree of keratopathy in a limbal stem cell deficient population. Methods: 37 patients (67 eyes) with aniridia-associated keratopathy (AAK) underwent corneal examination including slit lamp biomicroscopy to determine the Grade of AAK, Cochet-Bonnet esthesiometry, and in vivo confocal microscopy (IVCM) to assess morphology of the central corneal epithelium and subepithelial region. Results: AAK Grade ranged from 1 (limbal involvement only) to 4 (total conjunctivalization), with progression from Grade 1 occurring after the age of 20. 30% of subjects had an asymmetric Grade between eyes. In early-stage AAK (Grades 1-2), central epithelial cells had mixed corneal-conjunctival phenotype, touch sensitivity and subbasal nerves diminished, and mature dendritic cells, inflammatory leukocytes, and blood vessels were present despite central transparency in the slit lamp. In later stages (Grades 3-4) of the LSCD, neural deficit and nerve function worsened, immune cell invasion increased, and lymphatic vessels were detected in several cases. Goblet cells and epithelial cysts were observed to varying degrees in all stages, but without clear association to AAK severity. The clinical grade and progression of AAK was strongly associated with the central corneal epithelial phenotype. Conclusions: AAK is associated with degradation of epithelial phenotype, a neural deficit, and immune compromised status even in the clear central cornea in the earliest stages. IVCM can aid in assessing whether the conditions for limbal stem cell maintenance are likely to exist, based on morphology of the central epithelial microenvironment. (c) 2017 The Authors. Published by Elsevier Inc.

Place, publisher, year, edition, pages
ELSEVIER SCIENCE BV , 2018. Vol. 16, no 1, p. 163-172
Keywords [en]
Aniridia; Limbal stem cell deficiency; In vivo confocal microscopy; Keratopathy
National Category
Immunology
Identifiers
URN: urn:nbn:se:liu:diva-144451DOI: 10.1016/j.jtos.2017.11.003ISI: 000419225000018PubMedID: 29133179OAI: oai:DiVA.org:liu-144451DiVA, id: diva2:1176577
Note

Funding Agencies|Aniridia Norway Foundation; Region Ostergotland in Sweden

Available from: 2018-01-22 Created: 2018-01-22 Last updated: 2018-02-01

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Lagali, NeilFagerholm, Per
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Division of Neuro and Inflammation ScienceFaculty of Medicine and Health SciencesDepartment of Ophthalmology in Linköping
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