To report three cases of rituximab treatment in refractory orbital inflammatory disease.

Rituximab treatment was performed in three patients (thyroid-associated ophthalmopathy, orbital myositis, and idiopathic sclerosing inflammation) unresponsive to corticosteroid and radiotherapy. One cycle consisted of intravenous infusion of 1000 mg of rituximab, twice at a two-week interval. The patient with thyroid-associated ophthalmopathy exhibited clinical improvement in visual acuity, color vision, visual evoked potential, and visual field after one cycle of rituximab treatment. The patient with orbital myositis improved showed diplopia with a decrease in the size of the extraocular muscle after two cycles of treatment. The patient with idiopathic sclerosing inflammation showed improvements in visual acuity and visual field defect after one cycle of therapy. There were no severe infections or hematologic adverse effects including neutropenia, or decreased immunoglobulin during the follow-up period.

Treatment with rituximab appears to offer another therapeutic option in patients unresponsive to corticosteroid or radiotherapy.