I report a case with ocular Behçet disease presenting as hypopyon-anterior uveitis refractory to conventional immunosuppressive drugs who was safely and effectively managed for 20 years by nonstop infusions of a modern anti-TNF agent, infliximab. A 14-year-old girl who had a history of recurrent oro-genital ulcers, arthralgia, and pathergy test positivity presented with the symptoms of bilateral blurred vision, ocular pain, photophobia, and lacrimation. Initial visual acuities were 20/25 bilaterally, and biomicroscopy revealed bilateral iridocyclitis with left shifting cold hypopyon formation in the anterior chamber of the eye. The diagnosis of ocular Behçet disease was made, and topical anti-inflammatory drops were initiated with a cycloplegic agent. In due course, the child complained of chronic floaters and decreased vision from 20/25 to 20/200 bilaterally. Vitreitis, retinitis, perivasculitis, and cystoid macular edema (CME) were encountered, and the combination of corticosteroid, azathioprine, and cyclosporine-A was immediately started. However, the child did not respond to this conventional management for 3 months. A prompt and dramatic response was obtained with repeated Remicade® (infliximab) infusions (weeks 0, 2, 6, and 10), which led to fast improvement in her systemic and ocular symptoms. Long-term remission of 20 years was obtained by regular infliximab infusions at each 6- to 9-week interval with stabilized vision (20/20) and resolution of CME. The present case shows the significant role of biologicals in pediatric severe panuveitis and that nonstop infliximab infusions initiated early led to rapid and long-term control of intraocular inflammation and recurrent sight-threatening uveitis attacks for protecting the vision in children with severe OBD.