Yue Tong, Yu Chengdong, Yan Yuchun, Chu Weihong, He Baoping, Kang Min, Xu Yingjie, Zhang Dan, Li Ming, Wen Min, Wu Feifei, Hou Jun, Su Gaixiu, Wu Fengqi, Lai Jianming, Zhu Jia
Department of Rheumatology and Immunology, Capital Center for Children's Health, Capital Medical University, Beijing, 100020, China.
Department of Growth and Development, Capital Institute of Pediatrics, Beijing, 100020, China.
Ital J Pediatr. 2025 Aug 20;51(1):257. doi: 10.1186/s13052-025-02091-8.
This multicenter study aimed to address the heterogeneity of chronic recurrent multifocal osteomyelitis (CRMO) by identifying clinical subtypes through cluster analysis, exploring clinical features, treatment approaches, and short-term prognosis to improve management of pediatric CRMO.
Data from 42 pediatric CRMO patients (47.6% male; mean age 7.87 ± 3.45 years) diagnosed between June 2018 and June 2024 were analyzed. Using cluster analysis with 17 variables, patients were categorized into phenotypic subgroups. Statistical tests assessed differences in clinical features, treatment, and outcomes. Kaplan-Meier survival analysis and log-rank tests evaluated recurrence risk and final Physician Global Assessment(PGA) scores.
Patients were classified into two groups: chronic bone pain and acute systemic inflammation. Significant differences were found in fever occurrence (P = 0.002), C-reactive protein (CRP), interleukin-6(IL-6), cytokines including tumor necrosis factor-α(TNF-α) elevation (P = 0.013, 0.003, 0.029), and Hemoglobin(HB), alkaline phosphatase (ALP) reduction (P = 0.007, < 0.001). PGA scores also differed significantly (P < 0.001). Although baseline differences existed, post-treatment recurrence risk and final PGA scores showed no significant differences (P = 0.247, P = 0.211). Treatment differed only in glucocorticoid use; non-steroidal anti-inflammatory drugs (NSAIDs), disease-modifying antirheumatic drugs(DMARDs), TNF inhibitors, and diphosphonates showed no statistical differences. Both groups reached remission approximately 12 months post-diagnosis.
Two distinct clinical phenotypes of pediatric CRMO were identified, each achieving favorable outcomes with tailored treatments. Recognizing these phenotypes may guide clinical strategies and improve prognosis for CRMO patients.
这项多中心研究旨在通过聚类分析确定临床亚型,探讨慢性复发性多灶性骨髓炎(CRMO)的临床特征、治疗方法和短期预后,以解决其异质性问题,从而改善儿童CRMO的管理。
分析了2018年6月至2024年6月期间诊断的42例儿童CRMO患者的数据(男性占47.6%;平均年龄7.87±3.45岁)。使用包含17个变量的聚类分析将患者分为表型亚组。统计检验评估临床特征、治疗和结果的差异。Kaplan-Meier生存分析和对数秩检验评估复发风险和最终医生整体评估(PGA)评分。
患者被分为两组:慢性骨痛组和急性全身炎症组。在发热发生率(P = 0.002)、C反应蛋白(CRP)、白细胞介素-6(IL-6)、包括肿瘤坏死因子-α(TNF-α)升高的细胞因子(P = 0.013、0.003、0.029)以及血红蛋白(HB)、碱性磷酸酶(ALP)降低方面存在显著差异(P = 0.007、<0.001)。PGA评分也有显著差异(P < 0.001)。虽然存在基线差异,但治疗后复发风险和最终PGA评分无显著差异(P = 0.247、P = 0.211)。治疗仅在糖皮质激素使用上有所不同;非甾体抗炎药(NSAIDs)、改善病情抗风湿药(DMARDs)、TNF抑制剂和双膦酸盐无统计学差异。两组在诊断后约12个月均达到缓解。
确定了儿童CRMO的两种不同临床表型,每种表型通过量身定制的治疗都能取得良好效果。认识到这些表型可能指导临床策略并改善CRMO患者的预后。