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生物治疗慢性复发性多灶性骨髓炎:4 例报告及文献复习。

Biologic treatment for chronic recurrent multifocal osteomyelitis: report of four cases and review of the literature.

机构信息

Pediatric Unit, Department of Medical and Surgical Sciences, S. Orsola-Malpighi Hospital, University of Bologna, Via Massarenti 9, Bologna, Italy.

Pediatric Radiology Unit, Sant'Orsola-Malpighi Hospital, University of Bologna, Bologna, Italy.

出版信息

Rheumatol Int. 2018 Jan;38(1):153-160. doi: 10.1007/s00296-017-3877-0. Epub 2017 Nov 11.

Abstract

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare non-infectious inflammatory disorder with unpredictable clinical course, characterized by acute exacerbations and spontaneous remissions. There are no randomized-controlled trials about treatment options. Non-steroidal anti-inflammatory drugs (NSAID) are the first-line treatment option; glucocorticoids seem to be effective; positive outcomes have been obtained with bisphosphonates. In the last few years successful use of biologic agents like anti-TNF agents has been reported. We report the cases of 3 children suffering from CRMO who were treated with NSAID, steroid, bisphosphonates and eventually received etanercept and 1 case without vertebral involvement treated with etanercept after NSAID and steroid; all cases showed clinical improvement. The mean ages at symptoms onset and diagnosis were 8 and 10 years and 10 months, respectively. Two patients presented with back pain and three had vertebral lesions. Mean interval from diagnosis to the onset of anti-TNF treatment was 14 months. According to our small experience, we suggest considering therapy with etanercept for the treatment of severe cases with persistently active disease despite multiple treatments.

摘要

慢性复发性多灶性骨髓炎(CRMO)是一种罕见的非传染性炎症性疾病,具有不可预测的临床病程,其特征为急性加重和自发性缓解。目前尚无关于治疗选择的随机对照试验。非甾体抗炎药(NSAID)是一线治疗选择;糖皮质激素似乎有效;双膦酸盐也取得了良好的效果。在过去几年中,已有报道称生物制剂(如抗 TNF 药物)的使用取得了成功。我们报告了 3 例患有 CRMO 的儿童,他们分别接受了 NSAID、类固醇、双膦酸盐治疗,最终接受了依那西普治疗,1 例无脊椎受累的儿童在 NSAID 和类固醇治疗后接受了依那西普治疗;所有病例均显示出临床改善。症状发作和诊断的平均年龄分别为 8 岁和 10 岁 10 个月。2 例患者表现为背痛,3 例患者有脊椎病变。从诊断到开始抗 TNF 治疗的平均间隔为 14 个月。根据我们的经验,我们建议对于尽管经过多种治疗仍持续活动的严重病例,考虑使用依那西普进行治疗。

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