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

Abstract

PurposeTo report a case of pediatric Vogt-Koyanagi-Harada (VKH) successfully treated with infliximab and methotrexate for ten years.ObservationsA 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 importanceCombined 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.

Successful treatment of adalimumab in a child with Vogt-Koyanagi-Harada: which is the best available systemic treatment?

“…Among the common disease-modifying anti-rheumatic drugs (DMARDs), 7 children received cyclosporine A, 4 azathioprine, 2 mycophenolate mofetil and 15 methotrexate (MTX) reporting the treatment as efficacy in 2/7 (28.6%), 0/4, 0/2, and 6/15 (40%) in monotherapy respectively (2-16). Among biologic drugs, 4 patients received infliximab with concomitant MTX describing an ocular response in 3/4 (75%) patients (3,6,14). Rituximab was administered in 2 patients with resolution of ocular inflammation in both patients (10,16).…”

Long-term recurrence interval of Vogt–Koyanagi–Harada disease post-COVID-19 vaccination: A case study

“…Vogt-Koyanagi-Harada (VKH) disease is a chronic and systemic in ammatory disorder characterized by bilateral panuveitis [1], which is associated with serous retinal detachments, disk edema, and vitritis, with eventual development of sunset glow fundus. Although not all patients present with the full constellation of these extraocular ndings [2], tinnitus, hearing loss, vertigo, meningismus, poliosis, and vitiligo may be associated with VKH.…”

Systemic glucocorticoid-free therapy with adalimumab plus immunosuppressants versus conventional therapy in treatment-naïve Vogt-Koyanagi-Harada disease patients

“…Research has shown that the level of TNF-α is elevated in patients who suffer from non-infectious uveitis, including VKH. Neutralizing TNF-α with TNF-α inhibitors has been shown to be a feasible tactic in the treatment of VKH in several case reports (9,(13)(14)(15)(16). Adalimumab (ADA, Humira, AbbVie, North Chicago, Illinois, USA), a fully humanized monoclonal antibody that specifically targets TNF-α, was shown to be an ideal glucocorticoid-sparing agent in a series of rheumatic diseases (17,18).…”