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Case Reports
. 2014 May 24:7:316.
doi: 10.1186/1756-0500-7-316.

Efficacy of adalimumab in young children with juvenile idiopathic arthritis and chronic uveitis: a case series

Affiliations
Case Reports

Efficacy of adalimumab in young children with juvenile idiopathic arthritis and chronic uveitis: a case series

Francesco La Torre et al. BMC Res Notes. .

Abstract

Background: Juvenile idiopathic arthritis is a relatively common chronic disease of childhood, and is associated with persistent morbidity and extra-articular complications, one of the most common being uveitis. The introduction of biologic therapies, particularly those blocking the inflammatory mediator tumor necrosis factor-α, provided a new treatment option for juvenile idiopathic arthritis patients who were refractory to standard therapy such as non-steroidal anti-inflammatory drugs, corticosteroids and/or methotrexate.

Case presentations: The first case was a 2-year-old girl with juvenile idiopathic arthritis and uveitis who failed to respond to treatment with anti-inflammatories, low-dose corticosteroids and methotrexate, and had growth retardation. Adalimumab 24 mg/m2 every 2 weeks and prednisone 0.5 mg/kg/day were added to methotrexate therapy; steroid tapering and withdrawal started after 1 month. After 2 months the patient showed good control of articular and ocular manifestations, and she remained in remission for 1 year, receiving adalimumab and methotrexate with no side effects, and showing significant improvement in growth. Case 2 was a 9-year-old boy with an 8-year history of juvenile idiopathic arthritis and uveitis that initially responded to infliximab, but relapse occurred after 2 years off therapy. After switching to adalimumab, and adjusting doses of both adalimumab and methotrexate based on body surface area, the patient showed good response and corticosteroids were tapered and withdrawn after 6 months; the patient remained in remission taking adalimumab and methotrexate. The final case was a 5-year-old girl with juvenile idiopathic arthritis for whom adalimumab was added to methotrexate therapy after three flares of uveitis. The patient had two subsequent episodes of uveitis that responded well to local therapy, but was then free of both juvenile idiopathic arthritis and uveitis symptoms, allowing methotrexate and then adalimumab to be stopped; the patient remained in drug-free remission.

Conclusion: This report includes the first published case of the use of adalimumab in a child aged <3 years. Our clinical experience further supports the use of biologic therapy for the management of juvenile idiopathic arthritis and uveitis in children as young as two years of age.

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Figures

Figure 1
Figure 1
Case 1 left knee ultrasound. Transverse gray-scale and color Doppler 12–5 MHz ultrasound image obtained over the medial aspect of the knee showing abundant synovial pannus filling the subquadriceps recess as a band of hypoechoic tissue intermingled with fluid. Marked synovial hyperemia is observed at color Doppler examination indicating active pannus.
Figure 2
Figure 2
Case 1 left ankle ultrasound. Midsagittal 12–5 MHz ultrasound image over the dorsal ankle shows a distended anterior joint recess, filled with hypoechoic fluid with active synovial pannus as marked by color Doppler examination.
Figure 3
Figure 3
Case 1 (age 2 years). Photograph patient with juvenile idiopathic arthritis with fixed flexion contraction of the left leg for active knee and ankle joints arthritis (red arrows).
Figure 4
Figure 4
Case 1 left knee ultrasound after treatment. Left knee: transverse gray-scale 12–5 MHz ultrasound image obtained over the medial aspect of the knee showing normal aspect of the joint without fluid or synovial pannus into the subquadriceps recess.
Figure 5
Figure 5
Case 1 left ankle ultrasound after treatment. Midsagittal12 − 5 MHz US image over the dorsal ankle showing normal joint recess, without hypoechoic fluid or synovial pannus.
Figure 6
Figure 6
Case 1 after 2 years of ongoing adalimumab treatment. Photograph showing patient in remission at 4 years of age.

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