Journal List > J Korean Ophthalmol Soc > v.48(11) > 1007972

Park, Wee, Lee, and Kim: A Case of Idiopathic T-cell Mediated Chronic Conjunctivitis With Limbitis Treated Using Oral Cyclosporine

Abstract

Purpose

To report a case of refractory idiopathic T cell mediated chronic conjunctivitis causing limbal insufficiency, which improved with cyclosporine therapy.

Case Summary

A 43-year old man complained of conjunctival injection and discomfort in both eyes that lasted three years and was refractory to topical steroids, antibiotics, and artificial tears. There was no evidence of connective tissue diseases, Stevens-Johnson syndrome, pemphigoid, or drug history. Both eyes presented with diffuse injection and thickening of the conjunctiva, punctuated epithelial erosion of the conjunctiva and cornea, severe limbal epithelitis and elevated intraocular pressure. The left cornea was conjunctivalized due to limbal deficiency. Histological examination revealed severe infiltration of T and B cells, without any evidence of tumor cells or basement membrane anomaly. When treated with oral cyclosporine, injection and thickening of the conjunctiva decreased and the intraocular pressure was normalized. The surface inflammation of both eyes completely resolved two months after the treatment was initiated.

Conclusions

In chronic idiopathic conjunctivitis with limbitis resistant to conventional treatment and T cell infiltration found in pathological examination, oral cyclosporine therapy might be required to resolve inflammation.

References

1. Lindquist TD. Conjunctivits: An overview and classification. Krachmer JH, Mannis MJ, Holland EJ, editors. Cornea. 2nd ed.USA: Mosby;2005. v. 1. chap. 48.
2. Lee KJ, Ko CH, Kang YK, Lee DH. A case of bilateral conjunctival malignant lymphoma misdiagnosed as allergic conjunctivitis. J Korean Ophthalmol Soc. 1999; 40:2313–8.
3. Sohn SW, Park SK, Roh JH. One case of sebaceous carcinoma than masquerades as a chronic unilateral blepharoconjunctivits. J Korean Ophthalmol Soc. 2000; 41:521–5.
4. Stevenson D, Tauber J, Reis BL. Efficacy and safety of cyclosporin A ophthalmic emulsion in the treatment of moderate-to-severe dry eye disease: a dose-ranging, randomized trial. The Cyclosporin A Phase 2 Study Group. Ophthalmology. 2000; 107:967–74.
5. Sall K, Stevenson OD, Mundorf TK, Reis BL. Two multicenter, randomized studies of the efficacy and safety of cyclosporin ophthalmic emulsion in moderate to severe dry eye disease. CsA Phase 3 Study Group. Ophthalmology. 2000; 107:631–9.
6. Avunduk AM, Avunduk MC, Erdol H, et al. Cyclosporin effects on clinical findings and impression cytology specimens in severe vernal keratoconjunctivitis. Ophthalmologica. 2001; 215:290–3.
7. Akpek EK, Dart JK, Watson S, et al. A randomized trial of topical Cyclosporine 0.05% in topical steroid-resistant atopic keratoconjunctivitis. Ophthalmology. 2004; 111:476–82.
8. Sall K, Stevenson OD, Mundorf TK, Reis BL. the CsA Phase 3 Study Group. Two, multicenter, randomized studies of the efficiency and safety of cyclosporine ophthalmic emulsion in moderate to severe dry eye disease. Ophthalmology. 2000; 107:631–9.
9. Hoang-Xuan T, Prisant O, Hannouche D, Robin H. Systemic Cyclosporin A in severe atopic keratoconjunctivitis. Ophthalmology. 1997; 104:1300–5.
10. Faraj HG, Hoang-Xuan T. Chronic cicatrizing conjunctivitis. Curr Opin Ophthalmol. 2001; 12:250–7.
crossref
11. Ahmed M, Zein G, Khawaja F, Foster CS. Ocular cicatricial pemphigoid: pathogenesis, diagnosis and treatment. Prog Retin Eye Res. 2004; 23:579–92.
crossref
12. Mondino BJ, Brown SI. Immunosuppressive therapy in ocular cicatricial pemphigoid. Am J Ophthalmol. 1983; 96:453–9.
crossref
13. Renfro L, Snow JS. Ocular effects of topical and systemic steroids. Dermatol Clin. 1992; 10:505–12.
crossref
14. Miserocchi E, Baltatzis S, Roque MR, et al. The effect of treatment and its related side effects in patients with severe ocular cicatricial pemphigoid. Ophthalmology. 2002; 109:111–8.
crossref
15. Rao SN, Rao RD. Efficacy of topical cyclosporine 0.05% in the treatment of dry eye associated with graft versus host disease. Cornea. 2006; 25:674–8.
crossref
16. Den S, Sotozono C, Kinoshita S, Ikeda T. Efficacy of early systemic betamethasone or cyclosporin A after corneal alkali injury via inflammatory cytokine reduction. Acta Ophthalmol Scand. 2004; 82:195–9.
crossref
17. Althaus C, Daqres E, Reinhard T, et al. Cyclosporin-A and its metabolites in the anterior chamber after topical and systemic application as determined with high-performance liquid chromatography-electrospray mass spectrometry. Ger J Ophthalmol. 1996; 5:189–94.
18. Theng J, Zhou L, Tan D, La KW. Distribution of cyclosporin A in the cornea after topical or oral administration. J Ocul Pharmacol Ther. 2002; 18:83–8.
crossref
19. Calne RY, White DJ, Thiru S, et al. Cyclosporin A in patients receiving renal allografts from cadaver donors. Lancet. 1978; 2:1323–7.
crossref
20. Leonardi A, DeFranchis G, Fregona IA, et al. Effects of cyclosporin A on human conjunctival fibroblasts. Arch Ophthalmol. 2001; 119:1512–7.
crossref

Figure 1.
Conjunctival thickening, diffuse limbitis and diffuse punctuate epithelial erosion of the conjunctiva and cornea in the right eye (A and B). In the left eye, (C and D) conjunctivalization and diffuse limbal deficiency were accompanied with diffuse limbitis and conjunctivitis.
jkos-48-1567f1.tif
Figure 2.
The H&E staining (A) shows chronic inflammation with lymphoid cell infiltration, but there was no infiltration in the basement membrane or malignant cells. The immunohistochemistry staining against CD3 (B) and L26 (C) shows infiltration of T and B lymphocytes in the stroma. (×100)
jkos-48-1567f2.tif
Figure 3.
The conjunctival inflammation completely subsided in both eyes 10 weeks after he was treated with oral cyclosporine. The cornea became clear in the right eye (A and C) but the left eye showed stromal opacity and limbal neovascularization due to limbal insufficiency (B and D).
jkos-48-1567f3.tif
TOOLS
Similar articles