Bhat Chandrika S, Anderson Catriona, Harbinson Aoibhinn, McCann Liza J, Roderick Marion, Finn Adam, Davidson Joyce E, Ramanan Athimalaipet V
Departments of Paediatric Rheumatology and Immunology, Bristol Royal Hospital for Children, Bristol, BS2 8BJ, UK.
Scottish Paediatric and Adolescent Rheumatology Network, NHS National Services Scotland, Meridian Court, 5 Cadogan Street, Glasgow, G2 6QE, UK.
Pediatr Rheumatol Online J. 2018 Nov 22;16(1):74. doi: 10.1186/s12969-018-0290-5.
To understand the demographics, clinical features and treatment outcomes of Chronic Non-bacterial Osteitis (CNO) from three tertiary paediatric rheumatology services in the United Kingdom.
Children less than 18 years of age diagnosed with CNO between 2001 to 2016 from one tertiary service and between 2001 to 2017 from two tertiary services were included. Clinical notes were reviewed and all pertinent data were collected on a pre-defined proforma. One hundred and thirty one patients were included in the study. The Bristol diagnostic criteria were applied retrospectively.
Retrospective analysis of the data showed that the disease was more common in girls than boys (2.5:1), median age at onset of symptoms was 9.5 years (IQR 8 to 11 years). Bone pain was the predominant symptom in 118/129 (91.4%) followed by swelling in 50/102 (49.01%). Raised inflammatory markers were present in 39.68% of the patients. Whole body Magnetic Resonance Imaging (MRI) was a useful diagnostic tool. Metaphyses of long bones were most often involved and the distal tibial metaphyses 65/131 (49.6%) was the most common site. Non-steroidal anti-inflammatory drugs were used as first line (81.67%) followed by bisphosphonates (61.79%). Treatment was escalated to a TNF blocker when response to bisphosphonates was suboptimal. The disease was in remission in 82.4% of the patients during the last follow up.
Our multicentre study describes features and outcomes of CNO in a large number of patients in the United Kingdom.
Raised inflammatory markers were present in 39.68% of our patients. Whole body MRI is useful for diagnosis and also determining response to treatment. A greater number of lesions were detected on radiological imaging compared to clinical assessment. Metaphyses of long bones were most often involved and the distal tibial metaphyses (49.6%) were the most common site. Non-steroidal anti-inflammatory drugs were used as first line (81.67%) followed by bisphosphonates (61.79%). There was no difference in number of medications used for management in unifocal versus multifocal disease. TNF blockers were used with good effect in our cohort.
了解英国三家三级儿科风湿病服务机构中慢性非细菌性骨炎（CNO）的人口统计学特征、临床特征及治疗结果。
纳入2001年至2016年在一家三级服务机构以及2001年至2017年在另外两家三级服务机构中诊断为CNO的18岁以下儿童。回顾临床记录，并按照预先定义的表格收集所有相关数据。131例患者纳入研究。回顾性应用布里斯托尔诊断标准。
对数据的回顾性分析显示，该疾病在女孩中比男孩更常见（2.5:1），症状出现的中位年龄为9.5岁（四分位间距8至11岁）。118/129例（91.4%）患者以骨痛为主要症状，50/102例（49.01%）患者伴有肿胀。39.68%的患者炎症标志物升高。全身磁共振成像（MRI）是一种有用的诊断工具。长骨干骺端最常受累，胫骨干骺端远端65/131例（49.6%）是最常见部位。非甾体类抗炎药作为一线用药（81.67%），其次是双膦酸盐（61.79%）。当双膦酸盐治疗效果欠佳时，治疗升级为使用肿瘤坏死因子拮抗剂。在最后一次随访时，82.4%的患者病情缓解。
我们的多中心研究描述了英国大量CNO患者的特征和结局。
我们39.68%的患者炎症标志物升高。全身MRI对诊断及确定治疗反应均有用。与临床评估相比，影像学检查发现的病变更多。长骨干骺端最常受累，胫骨干骺端远端（49.6%）是最常见部位。非甾体类抗炎药作为一线用药（81.67%），其次是双膦酸盐（61.79%）。单灶性疾病与多灶性疾病在用药数量上无差异。在我们的队列中，肿瘤坏死因子拮抗剂使用效果良好。
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