摘要:

Purpose.We report a case of atypical bilateral interstitial keratitis associated with Cogan's syndrome.Methods.A 28-year-old man presented with a 2-year history of recurrent bilateral keratitis. Bilateral hearing loss preceded the ocular symptoms by 2 years. The patient also complained of skin nodules, headache, back pain, and arthritis. Corneal finding were consistent with superior stromal keratitis with stromal neovascularization and lipid deposition in the stroma. The patient's audiogram revealed cochlear pathology compatible with Cogan's syndrome (sensorineural deafness).Results.The patient was treated with topical steroids but eventually required corneal transplantation in the right eye as a consequence of progressive loss of vision secondary to progressive lipid keratopathy. Visual acuity at the patient's most recent follow-up evaluation was 20/40.Conclusion.This case represents an unusual type of interstitial keratitis associated with Cogan's disease. The absence of ocular symptoms at the time of initial ear involvement and the atypical presentation of the keratitis were responsible for the delay in diagnosis in this patient, resulting in hearing impairment.

ISSN:0277-3740    

出版商:OVID    

年代:2001

数据来源: OVID

ISSN:0277-3740    

出版商:OVID    

年代:2001

数据来源: OVID

摘要:

Purpose.We report clinical and histopathologic findings of a conjunctival lesion associated with xeroderma pigmentosum.Methods.A Saudi girl with known xeroderma pigmentosum presented with pain and photophobia of the right eye and an elevated temporally located perilimbal mass.Results.The mass was resected successfully and has not recurred during 1-year follow-up. Histopathologic examination of the tissue showed a necrobiotic granuloma with associated histiocytic infiltration. The patient had no systemic disease, lipid was not detected in the histiocytic lesion, no Touton giant cells were present, and there was no evidence of elastolysis.Conclusion.Ocular malignancies occur in association with xeroderma pigmentosum, but benign lesions that mimic a malignancy may occur.

ISSN:0277-3740    

出版商:OVID    

年代:2001

数据来源: OVID

ISSN:0277-3740    

出版商:OVID    

年代:2001

数据来源: OVID

摘要:

Purpose.To report the features of a syndrome of endothelial failure and band-shaped keratopathy in an infant with corpus callosum agenesis.Methods.The clinical and histopathologic features of an infant presenting with bilateral corneal clouding and corpus callosum agenesis are reported. The patient underwent bilateral penetrating keratoplasty at ages 28 months and 4 years. Light and electron microscopy were used to characterize the structural changes.Results.The epithelium was thin and degenerate. Bowman's membrane contained spherical aggregates that were present also within a connective tissue pannus. The midstroma was normal, but there were floral and rope-like aggregations of collagen in the pre-Descemet's membrane region. Under electron microscopy, the spherules formed target-shaped lesions with a central focus of alternating electron-dense and lucent material. Numerous microfilaments in the pannus and anterior stroma labeled with fibrillin-1 antibody. Microfilaments within fibroblasts were stained with vimentin antibody. Posteriorly, the endothelium was mainly absent and Descemet's membrane showed a fetal layer and a posterior collagenous layer.Conclusion.Corneal appearances in this patient were in keeping with those of congenital hereditary endothelial corneal dystrophy. However, there was no family history and neither parent showed a clinical endothelial abnormality. The presence of fetal, banded material in Descemet's membrane suggested that endothelial loss began at or near the time of birth. The band keratopathy was regarded as a secondary change. The association with corpus callosum agenesis does not appear to have been described previously.

ISSN:0277-3740    

出版商:OVID    

年代:2001

数据来源: OVID

摘要:

Purpose.To document the clinical and histopathologic corneal features of a patient who developed multifocal corneal argyrosis after a chemical explosion injury with unusual involvement of the corneal stroma and keratocytes.Methods.The corneal button was investigated by light and transmission electron microscopy and scanning electron microscopy combined with energy-dispersive x-ray microanalysis.Results.Clinically, the patient showed dark discoloration of the lids, periocular skin, episclera, and conjunctiva and had multiple brown dots in the superficial layers of the cornea. Microscopic examination of the cornea showed diffuse deposition of silver particles in the epithelial basement membrane, Bowman's layer, and Descemet's membrane. In the corneal stroma, silver granules accumulated intracellularly within lysosomal structures of degenerative keratocytes and extracellularly in association with collagen fibers and cellular debris. Energy-dispersive x-ray analysis showed peaks of silver and sulfur.Conclusion.The toxic influence of intracellular accumulation of silver in stromal keratocytes may lead to cell damage and necrosis and result in visual impairment.

ISSN:0277-3740    

出版商:OVID    

年代:2001

数据来源: OVID

ISSN:0277-3740    

出版商:OVID    

年代:2001

数据来源: OVID