Rheumatology Department, Princess Margaret Hospital for Children, Perth.
Murdoch Childrens Research Institute, Parkville and Department of Paediatrics, University of Melbourne, Victoria, Australia.
Rheumatology (Oxford). 2015 Sep;54(9):1688-91. doi: 10.1093/rheumatology/kev013. Epub 2015 May 3.
To describe the clinical features, management and outcome of 34 children with chronic recurrent multifocal osteomyelitis (CRMO) diagnosed at a single centre over 9 years.
All children identified with CRMO for the period 2005-13 were identified from a prospectively collected database, with additional data from hospital records.
Thirty-four patients, 21 female and 13 male, were identified. The average age at symptom onset was 9.8 years (range 3.8-17.9) and at diagnosis was 10.9 years (range 5.2-18.2), with an average delay in diagnosis of 12 months. Follow-up was 0.3-7.9 years (average 2.1), with 104 individual bony lesions identified, with a median of 3 (range 1-9) per patient. Six patients had unifocal disease. The sites involved included the tibia (n = 19), femur (n = 14), clavicle (n = 12), vertebrae (n = 10) and fibula (n = 8). Approximately half of patients had an inflammatory arthritis at diagnosis, and two-thirds in total eventually developed an arthritis. Pustulosis occurred in eight patients (24%), severe acne in four (12%) and psoriasis in three (9%). NSAIDs were used in 91%, CSs in 82% and MTX in 38%. Two patients were treated with anti-TNF agents. Episodic disease was most common (79%), while 21% had a monophasic pattern. Clinical remission occurred in 94% of children, with prolonged remission in 17%. Seven patients did not require medications for >12 months.
CRMO is more common than previously recognized, but diagnosis may be delayed. Episodic multifocal disease was most common, but some had unifocal and/or monophasic disease. Most patients responded to NSAIDs and/or intermittent CSs, but many required DMARDs.
描述在单一中心经过 9 年的时间诊断出的 34 例慢性复发性多灶性骨髓炎（CRMO）患儿的临床特征、治疗方法和转归。
从一个前瞻性收集的数据库中确定了 2005 年至 2013 年期间患有 CRMO 的所有儿童，同时从医院记录中获取了额外的数据。
共发现 34 名患者，其中 21 名女性，13 名男性。症状发作的平均年龄为 9.8 岁（范围 3.8-17.9），诊断时的平均年龄为 10.9 岁（范围 5.2-18.2），平均诊断延迟 12 个月。随访时间为 0.3-7.9 年（平均 2.1），共发现 104 个单独的骨病变，每个患者的中位数为 3（范围 1-9）。6 例为单灶性疾病。受累部位包括胫骨（n=19）、股骨（n=14）、锁骨（n=12）、椎体（n=10）和腓骨（n=8）。大约一半的患者在诊断时患有炎症性关节炎，而总共三分之二的患者最终发展为关节炎。8 例（24%）患者出现脓疱病，4 例（12%）患者出现严重痤疮，3 例（9%）患者出现银屑病。91%的患者使用了非甾体抗炎药（NSAIDs），82%的患者使用了皮质类固醇（CSs），38%的患者使用了甲氨蝶呤（MTX）。2 例患者使用了抗 TNF 药物。发作性疾病最为常见（79%），而单相型疾病占 21%。94%的患儿临床缓解，17%的患儿缓解持续时间延长。7 例患儿>12 个月无需药物治疗。
CRMO 比以前认为的更为常见，但可能会延迟诊断。发作性多灶性疾病最为常见，但也有一些为单灶性和/或单相性疾病。大多数患者对 NSAIDs 和/或间歇性 CSs 有反应，但许多患者需要 DMARDs。
