Rituximab for peripheral ulcerative keratitis with wegener granulomatosis.
OBJECTIVE
To demonstrate the effectiveness of Rituximab in the management of peripheral ulcerative keratitis (PUK) associated with Wegener granulomatosis (WG).


METHODS
A 50-year-old female with WG, confirmed by skin biopsy and positive anti-neutrophil cytoplasmic antibodies 3 years prior, was presented with a corneoscleral granulomatous mass that affected the superior limbus and with vasculitic PUK in the right eye. The mass was treated daily with oral prednisone (1 mg.kg-1.d-1) in conjunction with weekly maintenance treatment of 10 mg of methotrexate. After 2 months of treatment, the volume of the nodular granulomatous lesion decreased. However, the PUK and corneal thinning persisted, which presented a risk for corneal perforation. Subsequently, two 1000-mg infusions of rituximab were administered at weekly intervals.


RESULTS
One week after the first administration of rituximab, we observed persistence of corneal thinning, flattening of the nodular lesion, and disappearance of the necrotic foci. After the second rituximab treatment, the patient exhibited no signs of corneal perforation and we further observed total epithelialization of the PUK. No systemic side effects were seen. After 2 months, no nodular lesion was observed, although conjunctival epithelialization from the previous PUK remained. Weekly methotrexate and 10 mg of prednisone per day were maintained.


CONCLUSIONS
Rituximab seems to offer therapeutic promise in the treatment of refractory PUK associated with WG. Rituximab may be the elective treatment for severe anterior ocular inflammation associated with risk for corneal perforation, as a result of WG.
Huerva
Valentín
V
Department of Ophthalmology, University Hospital Arnau de Vilanova, Lleida, Spain. vhuerva@gmail.com
Sanchez
María Carmen
MC
Traveset
Alicia
A
Jurjo
Carmen
C
Ruiz
Agustín
A
D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D058846:Antibodies, Monoclonal, Murine-Derived; D018951:Antigens, CD20 / Q000276:immunology; D003320:Corneal Ulcer / Q000175:diagnosis / Q000188:drug therapy*; D004359:Drug Therapy, Combination; D005260:Female; D014890:Granulomatosis with Polyangiitis / Q000175:diagnosis / Q000188:drug therapy*; D006801:Humans; D007155:Immunologic Factors / Q000627:therapeutic use*; D008727:Methotrexate / Q000627:therapeutic use; D008875:Middle Aged; D011241:Prednisone / Q000627:therapeutic use; D000069283:Rituximab
D000911:Antibodies, Monoclonal; D058846:Antibodies, Monoclonal, Murine-Derived; D018951:Antigens, CD20; D007155:Immunologic Factors; D000069283:Rituximab; D011241:Prednisone; D008727:Methotrexate