Phenotypic Investigation of Regenerated Epithelial Cells After Gonococcal Corneal Perforation: A Clinical, Histological, and Immunohistochemical Study.

PURPOSE: To determine the characteristics of regenerated epithelial cells after severe gonococcal infection after corneal perforation. METHODS: Pathological tissue was obtained from the cornea at the time of surgery. Hematoxylin and eosin staining and immunohistochemical analysis were performed for cytoskeletal keratins (K12, K13, and K15), basement membrane and junctional markers (laminin 5, ZO-1 and Desmoplakin), and proliferative and mesenchymal markers (Ki67, α-SMA, and vimentin). RESULTS: A 42-year-old patient with severe gonococcal keratoconjunctivitis rapidly progressed to corneal perforation during administration of intensive topical and systemic antibiotics. After conservative treatment, the perforation healed and 5- × 3-mm corneal ectasia occurred with localized iris attachment. Complete closure of the cornea was confirmed by a negative Seidel test. After lamellar keratoplasty to improve corneal integrity and to prevent secondary glaucoma, the pathological tissue revealed a poorly organized epithelial layer at the regenerated ectatic area. The regenerated epithelial cells clearly expressed K12, ZO-1, and Desmoplakin with underlying laminin 5 (+) basement membrane. K15 and Ki67 expressions were observed predominantly at the limbal area but not in the regenerated area. α-SMA and vimentin were sporadically expressed in the underlying connective tissue. CONCLUSIONS: We speculate that the process of epithelial wound healing at the site of corneal perforation was responsible for migration of the surrounding epithelial cells. Although the regenerated cells expressed several cytokeratins and junctional markers, they remained disorganized and fragile.

Perforación corneal como primera manifestación de síndorme de Sjören secundario a artritis reumatoide / Corneal melts as the initial manifestation of sjögren syndrome secondary to rheumatoid arthritis

La artritis reumatoide es una enfermedad sistémica inflamatoria crónica frecuente, de etiología desconocida. El síndrome de Sjögren puede ir asociado a dicha patología. El curso clínico de la artritis reumatoide a nivel ocular es muy variable y el diagnóstico temprano es determinante para prevenir graves complicaciones. Presentamos el caso de una mujer de 64 años de edad que acudió al Servicio de Urgencias por presentar ojo rojo bilateral con fotofobia, lagrimeo continuo, dolor y disminución de la agudeza visual. En la exploración se objetivó importante adelgazamiento del estroma corneal en OD y perforación corneal OI, que requirió recubrimiento tectónico con membrana amniótica. Los análisis serológicos mostraron los siguientes resultados: Factor Reumatoide+, ANA+, ENA Anti-Ro/SSA+, ENA Anti-La/SSB+. La paciente fue diagnosticada de Artritis Reumatoide y Síndrome de Sjögren secundario. Desde entonces, sigue un tratamiento sistémico con corticoides y azatioprina. Aproximadamente, el 25% de los pacientes desarrollan enfermedad oftalmológica, esencialmente queratoconjuntivitis seca (25%), epiescleritis, escleritis y queratitis. Estas manifestaciones son en general poco severas, pero hay un pequeño porcentaje de pacientes, como el caso que aquí presentamos, que sufren una inflamación ocular grave y que sin tratamiento inmunosupresor precozmente instaurado, pueden desarrollar úlceras corneales estériles, centrales o periféricas, que les puede llevar incluso a la perforación y destrucción del globo (AU)

Rheumatoid arthritis(RA) is a common chronic inflammatory autoinmune disease, with unknown etiology. Approximately 11-31% of RA patients have secondary Sjögren´s syndrome. Ophthalmologic manifestations of these diseases can cause corneal scarring, ulceration, infection, and even perforation; thus, although the prognosis is good for most patients with Sjögren syndrome and ophthalmologic features, individuals with complications have much guarded prognosis. We report the case of a 65 years old woman with photophobia, pain, tearing and blurred vision in both eyes. Slit lamp exam showed an important corneal melting right eye, and corneal perforation left eye, which required amniotic membrane transplantation. The diagnosis was: Rheumatoid Arthritis and secondary Sjögren´s Syndrome. Azathioprine treatment was started in combination with oral steroids. Approximately 25% of patients have ocular involvement, and keratoconjunctivitis sicca is the most frequent ocular complication. Although the prognosis is good in most cases, an early diagnosis is necessary to avoid several complications (AU)

Corneal perforation due to limbal involvement in Sézary syndrome.

BACKGROUND: The majority of lymphomas involving the eye and ocular adnexa are B-cell lymphomas. Ocular involvement by T-cell lymphoma is rare. We report a case of corneal perforation due to direct involvement of the corneal limbus by lymphoma in a patient with Sézary syndrome. METHODS: A 58-year-old male with cutaneous T-cell lymphoma presented with painful, left-sided corneal ulceration, a dense infiltrate, severe epitheliopathy, hypopyon and a diffuse confluent dermatitis involving the lids. He had a history of Sézary syndrome. Despite maximal treatment, this severe ulcerative keratitis progressed to central corneal perforation. The eye was subsequently enucleated and submitted for histopathological examination. RESULTS: Histopathological examination confirmed corneal ulceration with perforation. There was an infiltrate of large atypical cells at the limbus, with tropism for the overlying epithelium. Immunohistochemical staining of these cells was positive for CD2, CD3, CD5, CD4 and CD7. Staining for CD8, CD30 and CD56 was negative. The appearances were those of an epitheliotrophic T-cell lymphoma, and were considered to represent spread from the patient's underlying Sézary syndrome. The patient died 2 months later from bronchopneumonia. CONCLUSIONS: Ocular involvement by cutaneous T-cell lymphoma usually occurs in advanced disease, and carries a poor prognosis. This patient was immunocompromised due to advanced malignancy, and there was a high suspicion of infection as the primary cause of corneal ulceration. This case highlights that it is important to consider direct tumour infiltration as an initiating or contributing factor for corneal ulceration in such patients.

Severe Arthrographis kalrae keratomycosis in an immunocompetent patient.

PURPOSE: To describe a severe case of keratomycosis caused by Arthrographis kalrae requiring repeated keratoplasty. METHODS: A 42-year-old otherwise healthy soft contact lens wearer developed a unilateral central corneal ulcer. Treatment with topical and systemic voriconazole is described. RESULTS: Repeated microbiological testing of ocular swabs (culture) initially yielded Candida albicans. Under treatment with topical clotrimazole, the ulcer progressed, and a corneal perforation required a keratoplasty à chaud. For prophylaxis, the patient received fluconazole systemically and continuous topical clotrimazole. However, in 2 weeks time, the mycotic infiltrates penetrated the corneal transplant and led to a second keratoplasty only 1 month after the first one. In the meantime, the microbiological analysis of the first keratoplasty revealed A. kalrae, which was sensitive to voriconazole. High-dose serum level-controlled systemic voriconazole and topical voriconazole were able to stabilize, but not eliminate the infection. About 1 year after the start of the voriconazole therapy, treatment had to be discontinued because of side effects. Mycotic infiltrates increased, and even an intracameral voriconazole injection could not prevent a third and a fourth keratoplasty. CONCLUSIONS: Ocular infection with A. kalrae is very rare. The microbiological differentiation of A. kalrae can be difficult. Because a broad spectrum of fungi is sensitive to voriconazole, the early topical and possibly systemic treatment is a reasonable therapeutic option when a mycotic infection of the eye is suspected, even before the causative fungus is identified.