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Bilateral necrotizing scleritis and peripheral ulcerative keratitis associated with granulomatosis with polyangiitis

Poster Details

First Author: S.Stanojlovic SERBIA

Co Author(s):    S. Arandjelovic   T. Kalezic   B. Dacic   S. Djuric     

Abstract Details

Purpose:

To report the clinical course and medical treatment of progressive necrotizing scleritis and peripheral ulcerative keratitis (PUK) occurring in both eyes simultaneously, in a patient with generalized form of granulomatosis with polyangiitis (GPA), after remission of pulmonary and renal manifestations was achieved.

Setting:

University Eye Hospital, Belgrade

Methods:

Observational case report.

Results:

A 61-year-old man with GPA developed bilateral rapidly progressive necrotizing scleritis and PUK. Acute exacerbation of ocular inflammation occurred during maintenance treatment with oral cyclophosphamide (CYP) and 3 months after the induction regimen with 6 CYP pulsed was given. The area of inflammation involved the entire anterior globe and peripheral cornea of both eyes. More than 90% of the surface area healed within 8 weeks following treatment with 3 pulsed doses of methylprednisolon in addition to oral CYP. Although the inflammation was arrested in both eyes, the advanced long-standing PUK in his right eye led to visual loss.

Conclusions:

Severe ocular inflammation associated with a generalized GPA may develop during remission of systemic disease, thus, additional anti-inflammatory and immunosuppressive therapy is required. Introduction of high-dose pulsed methylprednisolone, in addition to maintenance dose prednisone and increased oral CYP, arrested bilateral necrotizing scleritis and PUK in a patient with generalized GPA associated with ophthalmic inflammation refractory to CYP induction regimen. FINANCIAL DISCLOUSRE: NONE

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