Department of Paediatrics, Leuven University Hospitals, Leuven, Belgium.
Department of Radiology, Leuven University Hospitals, Leuven, Belgium.
Pediatr Rheumatol Online J. 2022 Jun 13;20(1):41. doi: 10.1186/s12969-022-00698-3.
To evaluate clinical characteristics, imaging findings, therapeutic approach and outcome of paediatric patients with Chronic Non-Bacterial Osteomyelitis (CNO).
Retrospective review of 30 children diagnosed with CNO at two tertiary care centres in Belgium. Imaging data were evaluated by blinded paediatric radiologists.
Mean age at onset was 10.3 years and mean age at diagnosis was 11.7 years. Bone pain was the leading symptom (29/30 patients). Out of 180 symptomatic lesions, 131 were confirmed on MRI as hyperintense geographic lesions on STIR images at the metaphysis and epiphysis adjacent to growth plates of tubular bones. The most common sites of involvement were the lower limbs, spine, sternoclavicular joint and humerus. For nearly half of the patients (14/30) monotherapy with NSAIDs was sufficient to obtain remission. The remaining 16 patients received second-line therapy: bisphosphonates (n = 15/30), disease-modifying antirheumatic drugs (n = 7/30), etanercept (n = 4/30) and tocilizumab (n = 1/30). Remission was reached after a mean time of 37.6 months in 26/30 patients. The prognosis was worse for patients with spinal involvement, resulting in more long-term sequelae.
We present a multicentre paediatric cohort of 30 CNO patients. A typical pattern of bone involvement could be found on MRI. NSAIDs were administered as first-line treatment. Second-line strategies included bisphosphonates, corticosteroids, methotrexate, etanercept and tocilizumab.
Retrospectively registered. Registratienummer EC KUL: MP018023.
评估儿童慢性非细菌性骨髓炎（CNO）的临床特征、影像学表现、治疗方法和预后。
对比利时两家三级医疗中心的 30 名 CNO 患儿进行回顾性分析。由小儿放射科医生对影像学数据进行盲法评估。
发病时的平均年龄为 10.3 岁，确诊时的平均年龄为 11.7 岁。骨痛是最主要的症状（29/30 例患者）。180 处有症状病变中，131 处经 MRI 证实为管状骨干骺端和骨骺附近的 STIR 图像上呈高信号的局灶性病变。最常见的受累部位是下肢、脊柱、胸锁关节和肱骨。将近一半的患者（14/30）仅用非甾体抗炎药（NSAIDs）单药治疗即可缓解。其余 16 例患者接受二线治疗：双膦酸盐（n=15/30）、改善病情的抗风湿药（n=7/30）、依那西普（n=4/30）和托珠单抗（n=1/30）。26/30 例患者在平均 37.6 个月后达到缓解。有脊柱受累的患者预后较差，导致更多的长期后遗症。
我们报告了一个由 30 例 CNO 患儿组成的多中心儿科队列。在 MRI 上可以发现典型的骨受累模式。NSAIDs 被作为一线治疗药物。二线策略包括双膦酸盐、皮质类固醇、甲氨蝶呤、依那西普和托珠单抗。
回顾性注册。注册号 EC KUL：MP018023。
