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Corneal Endothelial Cell Abnormalities in an Early Stage of the Iridocorneal Endothelial Syndrome

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Corneal Endothelial Cell Abnormalities in an Early Stage of the Iridocorneal Endothelial Syndrome 624 BritishJournalofOphthalmology 1994; 78: 624-631 ORIGINAL ARTICLES - Laboratory science Br J Ophthalmol: first published as 10.1136/bjo.78.8.624 on 1 August 1994. Downloaded from Corneal endothelial cell abnormalities in an early stage of the iridocorneal endothelial syndrome W R Lee, G E Marshall, C M Kirkness Abstract appearance with a central highlight and a light A corneal disc, obtained from a 52-year-old peripheral surround.2-5 The presence of ICE cells woman suffering from an early stage of the can herald (a) corneal oedema with a normal IOP iridocorneal endothelial syndrome (ICE), was and band-shaped keratopathy, (b) glaucoma investigated by various morphological tech- caused by ICE cell migration across the niques to analyse the structural variations trabecular meshwork (Chandler's syndrome), in the endothelial cells and to identify the (c) glaucoma associated with iris atrophy (essen- collagen types within the abnormal layer of tial iris atrophy), or (d) iris melanocytic neoplasia Descemet's membrane. Scanning electron (iris naevus syndrome).'1 It is clear, however, microscopy of the posterior corneal surface that a spectrum of disease exists and many revealed a mosaic of (a) flat hexagonal cells patients exhibit features of more than one of the resembling irregular but normal endothelial classically described syndromes. cells, and (b) rounded hexagonal (ICE) cells In pathological material, endothelial mor- with numerous surface microvilli. Degenera- phology has been inconsistent. By light micro- tive changes were present in each cell type, but scopy, the endothelial cell population is depleted were more common in the flat hexagonal cells in some cases, particularly in Chandler's syn- which contained intracytoplasmic spaces. By drome,"8 and occasionally multilayered,9 transmission electron microscopy the flat hex- although in the majority of specimens the cells agonal cells exhibited many of the features of have formed a degenerate monolayer.90' By scan- normal endothelial cells in terms of organelles ning electron microscopy, the presence of and intercellular attachments, but lateral abundant surface microvilli over hemispherical invaginations were absent. The ICE cells cells and cytoplasmic blebbing have been the differed in that the apical surface was covered most noteworthy feature of ICE cells.'0 Flat http://bjo.bmj.com/ by microvilli and the cytoplasm contained hexagonal cells which varied considerably in size tonofilaments, which were also observed by and shape have also been recorded as a mosaic light microscopic immunocytochemical stain- or subpopulation on the posterior corneal ing. Most commonly, intercellular attach- surface.591I ments were rudimentary in both types of cell By transmission electron microscopy ICE cells and intercellular spaces were dilated, but have been shown to have endothelial cell desmosomes were sometimes prominent in the characteristics."' 12 By contrast, ICE cells have on September 28, 2021 by guest. Protected copyright. ICE cells where interdigitations were pro- been shown to have epithelial characteristics nounced. In some sectors, the basal surface of with desmosomes6 1213 and intracytoplasmic the ICE cells was indented by deposition of tonofilaments.68 1012 13 clumps of fibrillar collagenous material. An Descemet's membrane which normally con- immunocytochemical study of the abnormal tains two collagen types (IV and VIII)' 16 iS posterior deposits localised type IV collagen to thickened in ICE by the addition of a broad the amorphous matrix and collagen types III posterior collagenous layer (PCL)6IO'2 '7 occa- and V, but not type I, to the collagen fibril sionally containing wide banded collagen.9'7 bundles. Mononuclear inflammatory cells Small foci of fibrillar deposits behind the pos- were identified between the ICE cells in the terior non-banded zone were reported in one monolayer. The evidence suggests that some example of the early stage of the disease.'0 An University ofGlasgow of the flat hexagonal cells were undergoing a attempt to identify the collagen types in the Department of degenerative change while others were trans- PCL has not to our knowledge been attempted. Ophthalmology forming into ICE cells. In the present morphological study we describe W R Lee (Brj' Ophthalmol 1994; 78: 624631) the features of the endothelium in an early G E Marshall C M Kirkness uncomplicated case of the ICE syndrome in which, with the application of immunogold Department of The iridocorneal endothelial (ICE) syndrome is a immunocytochemistry, we have identified some Pathology rare W R Lee non-familial unilateral disorder which pro- of the constituents of the abnormal posterior G E Marshall gresses to glaucoma and is characterised clinic- collagenous layer which is secreted by the ICE Correspondence to: ally by a fine beaten silver appearance of all or cells. Professor W R Lee, Pathology part of the posterior corneal surface as seen by Department, Western Infirmary, Glasgow GIl 6NT. slit-lamp specular microscopy.'2 By endothelial Accepted for publication specular photomicroscopy, abnormal endo- Clinical history 8 February 1994 thelial cells (ICE cells) have a characteristic A female patient (born 10 May 1941) attended Cornealendothelial cell abnormalities in an early stage ofthe iridocorneal endothelial syndrome 625 the Tennent Institute in January 1993 with a Paraffin sections were stained with haema- unilateral decrease in visual acuity which was toxylin and eosin, periodic acid Schiff, and associated with some diurnal fluctuation in Masson and were also used for an immuno- Br J Ophthalmol: first published as 10.1136/bjo.78.8.624 on 1 August 1994. Downloaded from vision, the vision being worst in the morning and histochemical study of cytokeratins in the clearing slightly by mid-day. Such symptoms epithelium and the endothelium. The following had persisted for 5 years. There was no family antibodies were applied - CAM 5-2, AE 1/3, history ofcorneal disease. The referring ophthal- NCL, and MNF. The tissue for transmission mologist considered that the anterior chamber electron microscopy was processed through to was shallow and that a few peripheral anterior Araldite and semithin sections were stained with *synechiae were present. On examination toluidine blue. Ultrathin sections were examined the vision in the affected eye was reduced to in a Jeol 1200 EXII transmission electron micro- 6/24 with correction. There was stromal and scope. The quadrant used for scanning electron epithelial corneal oedema and a distorted microscopy was subject to critical point drying eccentric pupil with ectropion uveae. Fine endo- before gold coating and examination in a Jeol thelial detail was obscured by the oedema. No JSM 64000 scanning electron microscope. The other features were distinguishable and in block allocated for immunocytochemistry was particular the other eye was entirely normal. processed for London resin (LR) white embed- Intraocular pressure in both eyes was within ding. Details of the techniques for embedding, normal limits. Endothelial specular photomicro- antibody labelling, and immunocytochemistry scopy (ESP) was performed with considerable have been provided elsewhere. 145S difficulty because ofthe corneal oedema. In some areas, cells with central highlights were visible. Clinically this was felt to be ICE syndrome Results because of the unilaterality, the ESP appearance and the lack ofany clinical features suggestive of LIGHT MICROSCOPY posterior polymorphous dystrophy - namely, The epithelium was oedematous and the base- vesicles, snail track, and thickened Descemet's ment membrane was multilayered and infolded membrane. The small ectropion uveae was also in parts: there was no evidence of a band felt to be consistent with the ICE syndrome. keratopathy. Bowman's layer was intact and A corneal graft was performed on 12 January there were plentiful stromal keratocytes, but in 1993 and at follow up 9 months later the graft has one halfofthe cornea the lamellae appeared to be remained clear and the intraocular pressure has more widely dispersed owing to oedema. remained normal. Descemet's membrane measured 6 jim but there was evidence ofmultilayering in the PAS stained sections (Fig 1). The normal cuboidal endo- Materials and methods thelium was replaced either by vacuolated cells The keratoplasty disc measured 8 mm in or rounded cells and some cells were remarkably diameter and the stroma was cloudy. The speci- attenuated (Figs la, lb). Mononuclear inflam- http://bjo.bmj.com/ men was divided into four, and three parts were matory cells were present within the monolayer fixed in glutaraldehyde (2%) for paraffin histo- (Fig lb). Consistent results were obtained with logy, scanning electron microscopy, and conven- the immunohistochemical markers for cyto- tional transmission electron miscoscopy. The keratins. The rounded ICE cells stained remaining part was fixed in a paraformaldehyde positively while the flattened and vacuolated (4%)/glutaraldehyde (0-5%) mixture for 2 hours cells were weakly staining (Fig ld). The at room temperature for immunogold immuno- epithelium stained positively for each of the on September 28, 2021 by guest. Protected copyright. cytochemistry. 14 15 antibodies used. - .R .Lk..... .*. ....p.. ........... ........ .011.OffiftI III 11111.... i.'I. -.... T.T. Figure I Light micrographs ofthe posterior corneal surface. (a) Vacuoles lpq'- :% . (arrowheads) were a ,#& *Ai prominentfeature in some I A areas ofthe endothelium. (b) b In other sectors the cells
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