Purpose: To report a child with severe treatment resistant juvenile idiopathic arthritis associated uveitis (JIAU), a condition that typically presents as chronic anterior uveitis, although severe panuveitis with retinal vasculitis may rarely occur.Methods: We describe the clinical course, treatment response, and challenge, dechallenge, and rechallenge sequence in a child with severe refractory JIAU treated with combined infliximab and tofacitinib therapy.Results: The patient showed marked improvement in ocular inflammation, macular edema, and visual acuity following addition of tofacitinib to ongoing infliximab therapy. Inflammation recurred after holding infliximab despite continuation of tofacitinib and improved again following reintroduction of infliximab.Conclusion: This challenge, dechallenge, and rechallenge sequence suggests that neither infliximab nor tofacitinib alone provided adequate disease control, whereas their combination was associated with sustained suppression of ocular inflammation. Combined tumor necrosis factor alpha and Janus kinase inhibition may represent a rescue therapeutic option for carefully selected patients with severe refractory JIAU, although its long term safety and efficacy require further evaluation.