Long term treatment with infliximab in pediatric Vogt-Koyanagi-Harada disease

PURPOSE: To report a case of pediatric Vogt-Koyanagi-Harada (VKH) successfully treated with infliximab and methotrexate for ten years. OBSERVATIONS: A 9-year-old Hispanic girl with VKH disease, was successfully treated with oral methotrexate 15 mg/week and oral prednisone 40 mg/day (1mg/kg/day). But when oral prednisone was tapered to 10 mg/day over a 3-month period, inflammation recurred. Patient was considered as corticosteroid-dependent thus infliximab 7mg/kg/pulse was started on days 0, 15, 60 and every 60 days thereafter. Six months after, infliximab was increased to 10mg/kg/pulse as cells in the anterior chamber were still observed. After four months of treatment, ocular inflammation was fully controlled, oral prednisone was tapered to discontinuation over a period of 10 months and methotrexate was maintained at 15 mg/week. At 1-year follow up, infliximab was reduced to 6 mg/kg/pulse as patient remained stable on examination. After being treated for 3-years it was decided to discontinue infliximab however, 2 + anterior chamber cells recurred after a dose was skipped thus infliximab was restarted. After 10 years treatment with infliximab 6 mg/kg/pulse every 60 days and methotrexate 15 mg/week associated, no relapsing inflammatory episodes and resolution of physical features of Cushing's syndrome were observed. CONCLUSION AND IMPORTANCE: Combined therapy of infliximab and methotrexate for up to 10 years was efficacious in this girl in controlling recurrent inflammation without associated side effects. To the best of our knowledge, this is the longest reported clinical follow up of a pediatric VKH case supporting the use of infliximab and methotrexate without steroids treatment.