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Efficacy of tumour necrosis factor inhibitors in peripheral ulcerative keratitis in Granulomatosis with polyangiitis
Author(s) -
Verly E.,
De Kock J.,
Leroy B.P.,
Sys C.,
De Schryver I.
Publication year - 2017
Publication title -
acta ophthalmologica
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.534
H-Index - 87
eISSN - 1755-3768
pISSN - 1755-375X
DOI - 10.1111/j.1755-3768.2017.0t077
Subject(s) - medicine , infliximab , adalimumab , dermatology , granulomatosis with polyangiitis , etanercept , vasculitis , surgery , pathology , rheumatoid arthritis , tumor necrosis factor alpha , disease
Purpose To report the efficacy of TNF i (tumour necrosis factor inhibitor), as a treatment of therapy‐resistant peripheral ulcerative keratitis ( PUK ) in granulomatosis with polyangiitis ( GPA ), formerly known as Wegener granulomatosis. Methods Observational report about 2 cases, known with the diagnosis of GPA , presenting with therapy‐resistant PUK . Results 2 middle‐aged females, known with GPA were referred to our department with a history of chronic redness resistant to local corticosteroids. Both patients showed a unilateral limbal vasculitis with necrotizing keratitis on slit lamp examination. In the first case, the diagnosis of GPA was confirmed by positive anti‐neutrophil cytoplasmic antibodies ( ANCA ) and nasal septum biopsy. In the second case, the clinical presentation was considered as a viral keratitis due to recurrent unilateral inflammation. Polymerase chain reaction ( PCR ) could not confirm the viral aetiology. Extensive systemic evaluation revealed a cervical lymphadenopathy on PET ‐ CT scan. Serology was positive for ANCA . In both cases, the PUK was nonresponsive to systemic corticosteroids. In the first case, cyclophosphamide failed to control the inflammation. Because the risk of corneal perforation, a treatment with TNF i, respectively Infliximab and Adalimumab, were successfully initiated. Conclusions Potentially lethal, it is important to diagnose and treat GPA urgently. Recently, TNF i were reported to be effective in the treatment of therapy‐resistant GPA . Both patients were refractory to conventional immunomodulatory therapy. Infliximab and adalimumab successfully controlled the inflammation with disappearance of necrotic foci. In our experience, TNF i are a safe and effective therapeutic strategy.