Abstract

Abstract Background There is a need for additional treatment options in refractory JIA uveitis which has responded inadequately to MTX and anti TNFα. There has been interest in the use of tocilizumab, the APTITUDE trial using SC tocilizumab in JIA uveitis and the STOP-Uveitis study comparing 2 dosing regimens of iv tocilizumab in adults. Anecdotal evidence reports JIA uveitis patients who were stable on IV tocilizumab flaring when switched to sc administration. We describe the successful use of IV tocilizumab and concurrent sc MTX in two cases. Methods Retrospective review of patient paper and electronic medical records. Results Case 1.: A five-year-old female presented with severe anterior uveitis in her right eye. She was diagnosed with idiopathic uveitis and treated with topical steroid, oral then SC MTX infliximab, mycophenolate and adalimumab, all with inadequate control of her uveitis. At the age of 12, on adalimumab, she developed arthritis and her diagnosis was changed to JIA uveitis. 7 years after initial diagnosis she was commenced on IV tocilizumab 8mgs/kg 4-weekly with improved control of her uveitis although initially still requiring some topical steroid. She required intra-articular steroids for active arthritis and agreed to restart SC MTX. On tocilizumab and MTX combined, her arthritis settled and her topical eye drops were weaned for the first time since diagnosis. From age 5 until 14 she was continually on topical steroids, consequently developing raised intraocular pressure and a dense cataract. With improved disease control she had cataract surgery aged 14. She has maintained good disease control for the last five years on the combination of IV tocilizumab and SC MTX and has not required any further steroids. Case 2: A 3 year old female with oligoarticular JIA had severe bilateral uveitis at presentation. Initial treatment was with oral prednisolone, topical steroid and SC MTX. 6 months after diagnosis her uveitis remained active and she was commenced on infliximab with additional IV methylprednisolone. Venous access was challenging, required a portacath to facilitate treatment. By the age of 6 she had developed a cataract requiring surgery and still had incomplete control of her uveitis. Infliximab was increased to 10mgs/kg 4 weekly with little further benefit and at the age of 7 she was changed to adalimumab. On this she developed macular oedema requiring pulse IV methylprednisolone. 5 years after diagnosis she was commenced on IV tocilizimab initially 4 weekly, increasing after three months to 2 weekly. Her methylprednisolone was weaned and she has subsequently maintained good disease control on IV tocilizumab 10mgs/kg 2 weekly with SC methotrexate. Conclusion We describe two cases of refractory JIA and uveitis in whom IV tocilizumab with SC methotrexate has provided good disease control. Further studies are required to determine the optimal dosing regimen. Conflicts of Interest The authors declare no conflicts of interest.

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