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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.

DOI:10.1007/s00296-017-3877-0
PMID:29127574
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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J Rheumatol. 2017 Jul;44(7):1058-1065. doi: 10.3899/jrheum.161255. Epub 2017 May 1.
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Oral alendronate in pediatric chronic recurrent multifocal osteomyelitis.口服阿仑膦酸钠治疗儿童慢性复发性多灶性骨髓炎
Pediatr Int. 2017 Apr;59(4):506-508. doi: 10.1111/ped.13236.
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An Update on the Pathogenesis and Treatment of Chronic Recurrent Multifocal Osteomyelitis in Children.儿童慢性复发性多灶性骨髓炎发病机制与治疗的最新进展
锁骨慢性硬化性骨髓炎:锁骨切除术后的临床结果
Healthcare (Basel). 2024 Jan 15;12(2):202. doi: 10.3390/healthcare12020202.
4
Standardized reporting and quantification of whole-body MRI findings in children with chronic non-bacterial osteomyelitis treated with pamidronate.儿童慢性非细菌性骨髓炎经帕米膦酸二钠治疗后的全身 MRI 表现的标准化报告和定量分析。
Pediatr Rheumatol Online J. 2022 Oct 1;20(1):85. doi: 10.1186/s12969-022-00746-y.
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Chronic Nonbacterial Osteomyelitis in Children.儿童慢性非细菌性骨髓炎
Children (Basel). 2021 Jun 25;8(7):551. doi: 10.3390/children8070551.
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A complicated path to the CRMO diagnosis - case of a 9 year old girl whose story comes full circle.通往 CRMO 诊断的复杂之路——一位 9 岁女孩的病例,其故事圆满结束。
BMC Musculoskelet Disord. 2019 Aug 31;20(1):392. doi: 10.1186/s12891-019-2776-9.
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Chronic Recurrent Multifocal Osteomyelitis of the Hand: A Rare Pediatric Condition.手部慢性复发性多灶性骨髓炎:一种罕见的儿科疾病。
Hand (N Y). 2021 Mar;16(2):213-222. doi: 10.1177/1558944719846599. Epub 2019 May 29.
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Comparison of different treatment approaches of pediatric chronic non-bacterial osteomyelitis.比较儿童慢性非细菌性骨髓炎的不同治疗方法。
Rheumatol Int. 2019 Jan;39(1):89-96. doi: 10.1007/s00296-018-4151-9. Epub 2018 Aug 31.
Paediatr Drugs. 2017 Jun;19(3):165-172. doi: 10.1007/s40272-017-0226-4.
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Clinical and genetic association, radiological findings and response to biological therapy in seven children from Qatar with non-bacterial osteomyelitis.卡塔尔七名非细菌性骨髓炎患儿的临床与基因关联、影像学表现及生物治疗反应
Int J Rheum Dis. 2017 Sep;20(9):1286-1296. doi: 10.1111/1756-185X.12940. Epub 2016 Nov 9.
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[Non-bacterial chronic osteomyelitis: Experience in a tertiary hospital].[非细菌性慢性骨髓炎:一家三级医院的经验]
An Pediatr (Barc). 2016 Jul;85(1):18-25. doi: 10.1016/j.anpedi.2015.08.010. Epub 2015 Oct 23.
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Arthritis Rheumatol. 2015 Apr;67(4):1128-37. doi: 10.1002/art.39013.