Background: We report a rare adverse effect of rituximab, a monoclonal antibody against CD20 antigen targeted lymphocytes, which has been recently used in the potentially sight threatening recalcitrant non-infectious scleritis with promising results. It has been documented to have reduced the need of corticosteroids with minimal adverse effects. Case Report: A 25 year old female with recalcitrant scleritis due to granulomatosis with polyangiitis, not responding to systemic cyclosporine and steroid, presented with increased intraocular pressure following each of the three cycles of rituximab but complete resolution of scleritis. Conclusion: Hence, as the utility of rituximab expands, the awareness of rheumatologists and physicians regarding its ocular adverse effects is crucial.