Combined Infliximab and Tofacitinib Therapy for Severe Refractory Juvenile Idiopathic Arthritis Associated Panuveitis: A Challenge, Dechallenge, Rechallenge Case Report.

Abstract

Purpose: To report a child with severe treatment-resistant juvenile idiopathic arthritis-associated uveitis (JIAU), characterized by panuveitis, diffuse retinal vascular leakage, and refractory cystoid macular edema.

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 is consistent with a possible complementary effect of infliximab and tofacitinib in this patient. However, causality and pharmacologic synergy cannot be established from a single case. Combined tumor necrosis factor alpha and Janus kinase inhibition may represent an exceptional rescue approach for carefully selected patients with severe refractory JIAU, although its efficacy and long-term safety require further evaluation.