La Torre Francesco, Cattalini Marco, Teruzzi Barbara, Meini Antonella, Moramarco Fulvio, Iannone Florenzo
Department of Paediatrics, Antonio Perrino Hospital, Brindisi, Italy.
BMC Res Notes. 2014 May 24;7:316. doi: 10.1186/1756-0500-7-316.
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.
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and methotrexate based on body surface area, the patient showed good response and corticosteroids were tapered and withdrawn after 6 months; the patient remained in remission taking adalimumab and methotrexate.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.
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.
幼年特发性关节炎是儿童期相对常见的一种慢性病,与持续的发病及关节外并发症相关,其中最常见的并发症之一是葡萄膜炎。生物疗法的引入,尤其是那些阻断炎症介质肿瘤坏死因子-α的疗法,为对非甾体抗炎药、皮质类固醇和/或甲氨蝶呤等标准疗法难治的幼年特发性关节炎患者提供了一种新的治疗选择。
第一个病例是一名2岁女童,患有幼年特发性关节炎和葡萄膜炎,对抗炎药、低剂量皮质类固醇和甲氨蝶呤治疗无反应,且有生长发育迟缓。在甲氨蝶呤治疗基础上加用每2周一次的阿达木单抗24mg/m²和每日0.5mg/kg的泼尼松;1个月后开始逐渐减少并停用类固醇。2个月后,患者的关节和眼部表现得到良好控制,她维持缓解状态1年,接受阿达木单抗和甲氨蝶呤治疗且无副作用,生长情况有显著改善。病例2是一名9岁男孩,有8年幼年特发性关节炎和葡萄膜炎病史,最初对英夫利昔单抗有反应,但在停药2年后复发。改用阿达木单抗后,根据体表面积调整阿达木单抗和甲氨蝶呤的剂量,患者反应良好,6个月后逐渐减少并停用皮质类固醇;患者接受阿达木单抗和甲氨蝶呤治疗维持缓解状态。最后一个病例是一名5岁女童,患有幼年特发性关节炎,在葡萄膜炎三次发作后,在甲氨蝶呤治疗基础上加用阿达木单抗。该患者随后又有两次葡萄膜炎发作,对局部治疗反应良好,但此后幼年特发性关节炎和葡萄膜炎症状均消失,从而停用甲氨蝶呤,随后停用阿达木单抗;患者维持无药缓解状态。
本报告包括了首次发表的在3岁以下儿童中使用阿达木单抗的病例。我们的临床经验进一步支持生物疗法可用于治疗两岁及以上儿童的幼年特发性关节炎和葡萄膜炎。