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