Phlyctenular keratoconjunctivitis - an atypically severe case treated with systemic biologic immunosuppressive therapy.

Purpose: To report an atypically severe and refractory phlyctenular keratoconjunctivitis case treated successfully with systemic biologic immunosuppressive therapy. Methods: A 10-year-old female was followed in the ophthalmology clinic for three years for a severe form of bilateral PKC. The patient was treated for blepharitis and intestinal parasitosis, and underwent topical corticosteroid therapy, followed by subconjunctival injections and systemic corticosteroids with no clinical improvement. An association of topical cyclosporine A and oral methotrexate had no clinical response either. Phlyctenae of the cornea remained evident with neovascularization, progressive peripheral corneal thinning and occasional anterior chamber reaction. Results: The patient was treated with a combination of infliximab and methotrexate and corticosteroid therapy was tapered, with a fast and sustained resolution of the symptoms and corneal signs. Eleven months past initiation of the treatment, the patient remains asymptomatic and without any recurrence of the disease. Conclusion: Phlyctenular keratoconjunctivitis may present with a broad spectrum of symptoms and signs, and its severity varies significantly. In cases of severe PKC, which are refractory to conventional therapy, systemic biologic immunosuppressive therapy may be a valuable alternative.