Chronic non-bacterial osteomyelitis (CNO) is a group of immune-mediated diseases which appears in bone inflammation, destruction and some orthopaedic consequences, especially in the cases of spinal involvement. This study is to compare characteristics and treatment outcomes of CNO patients with spinal involvement. The retrospective cohort study included data from 91 pediatric patients with CNO. The diagnosis is based on Jannson’s criteria with morphological confirmation (nonspecific chronic inflammation). Spine involvement detected by X-ray, computed tomography, magnetic resonance imaging, and bone scan in 29 (31.9%) patients. No differences in the family history, concomitant immune-mediated diseases between spinal (SpCNO) and peripheral (pCNO) forms of CNO have been revealed. Only 5 (10.2%) SpCNO patients (10.2%) had monofocal monovertebral involvement. The main risk factors of spinal involvement were female sex: RR = 2.0 (1.1; 3.9), sensitivity (Se) = 0.66, specificity (Sp) = 0.6; multifocal involvement: RR = 2.1 (0.9; 5.0), Se = 0.83, Sp = 0.37; no foot bones involvement: RR = 3.1 (1.3; 7.5), Se = 0.83, Sp = 0.5; sternum involvement RR = 2.3 (1.3; 4.1), Se = 0.24, Sp = 0.94. In the linear regression analysis only female sex (p = 0.005), multifocal involvement (p = 0.000001) and absence of foot bones involvement (p = 0.000001) were independent risk factors of spinal involvement (p = 0.000001). The response rate on bisphosphonates and tumor necrosis factor-a inhibitors was 90.9% and 66.7%, consequently. Only 4/29 (13.8%) SpCNO patients underwent surgery due to severe spinal instability or deformities. The spinal involvement is frequent in CNO and could be crucial for choosing a treatment strategy. Bisphosphonates and TNFa-inhibitors could be effective treatment options for severe SpCNO.

中文翻译：

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儿童慢性非细菌性骨髓炎脊柱形式的特点和治疗结果：一项回顾性队列研究

慢性非细菌性骨髓炎（CNO）是一组免疫介导的疾病，特别是在脊柱受累的情况下，会出现在骨骼炎症，破坏和某些矫形外科后果中。本研究旨在比较CNO脊柱受累患者的特征和治疗结果。回顾性队列研究包括来自91名CNO患儿的数据。诊断基于詹恩森氏标准，并经形态学证实（非特异性慢性炎症）。X射线，计算机断层扫描，磁共振成像和骨扫描可检测到29名（31.9％）患者的脊柱受累情况。没有发现家族史的差异，脊柱（SpCNO）和周围（pCNO）形式的CNO之间存在伴随的免疫介导的疾病。只有5名（10.2％）SpCNO患者（10。2％）有单灶性单椎受累。脊柱受累的主要危险因素是女性：RR = 2.0（1.1; 3.9），敏感性（Se）= 0.66，特异性（Sp）= 0.6；多灶累及：RR = 2.1（0.9; 5.0），Se  = 0.83，Sp  = 0.37; 无脚骨受累：RR = 3.1（1.3; 7.5），Se  = 0.83，Sp  = 0.5; 胸骨受累RR = 2.3（1.3; 4.1），Se  = 0.24，Sp  = 0.94。在线性回归分析中，只有女性（p  = 0.005），多灶受累（p  = 0.000001）和没有脚骨受累（p  = 0.000001）是脊柱受累的独立危险因素（p = 0.000001）。因此，对双膦酸盐和肿瘤坏死因子-α抑制剂的反应率为90.9％和66.7％。由于严重的脊柱不稳或畸形，只有4/29（13.8％）的SpCNO患者接受了手术。脊髓受累在CNO中很常见，对于选择治疗策略可能至关重要。双膦酸盐和TNFa抑制剂可能是严重SpCNO的有效治疗选择。