Archawanantakul Khwanjira, Vilaiyuk Soamarat, Pakakasama Samart, Lerkvaleekul Butsabong
Department of Pediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand.
Division of Rheumatology, Department of Pediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, 270 Rama VI Rd, Ratchathewi, Bangkok, 10400, Thailand.
Pediatr Rheumatol Online J. 2025 Jul 15;23(1):73. doi: 10.1186/s12969-025-01121-3.
Musculoskeletal (MSK) symptoms are a frequent presentation in pediatric patients and may arise from a range of conditions, including juvenile idiopathic arthritis (JIA) and malignancies. Differentiating cancer-related MSK symptoms from JIA at initial presentation remains challenging due to overlapping clinical features. Delays in the diagnosis of malignancy can result in significant morbidity, underscoring the need for reliable diagnostic tools. The ONCOREUM score was developed to distinguish malignancies presenting with arthropathy from JIA and demonstrated high performance in initial validation. However, its utility is limited to patients with arthropathy and does not extend to other forms of MSK involvement. This study aimed to validate the ONCOREUM score in patients with arthropathy and to develop an expanded predictive model to distinguish cancer-related MSK symptoms from JIA.
Patients aged < 16 years diagnosed with cancer or JIA were included. This retrospective study was conducted in two phases: (1) Evaluating the ability of the ONCOREUM score to identify cancer with arthropathy, (2) Developing a model to differentiate cancer-related MSK symptoms from JIA using stepwise logistic regression analysis.
A total of 1,026 patients were reviewed (646 cancer, 380 JIA). In phase 1, 26 cancer patients (4.0%) and 351 JIA patients (92.4%) were included. The ONCOREUM score (cutoff = - 6) had a sensitivity of 88.5% and specificity of 65.0%, with an AUC of 0.77. In phase 2, MSK symptoms were present in 84 (13%) cancer cases (61 hematologic, 23 solid tumors). The best-fitting model was obtained through multivariable analysis: back pain (OR 15.58, 95% CI 2.77-87.64, p < 0.02), nocturnal pain (OR 789.97, 95% CI 51.26-12,175.54, p < 0.0001), limb bone pain (OR 24.11, 95% CI 6.91-84.12, p < 0.0001), pallor (OR 6.30, 95% CI 1.55-25.60, p < 0.01), morning stiffness (OR 0.03, 95% CI 0.002-0.57, p < 0.02), additive arthritis (OR 0.003, 95% CI 0.00-0.04, p < 0.0001), and monoarticular involvement (OR 0.02, 95% CI 0.00-0.23, p < 0.002). This model yielded an AUC of 0.99 (95% CI 0.98-0.99).
The refined predictive model is a promising clinical tool for differentiating cancer-associated MSK symptoms from JIA.
肌肉骨骼(MSK)症状在儿科患者中很常见,可能由多种疾病引起,包括幼年特发性关节炎(JIA)和恶性肿瘤。由于临床特征重叠,在初次就诊时将癌症相关的MSK症状与JIA区分开来仍然具有挑战性。恶性肿瘤诊断的延迟可能导致严重的发病率,这突出了对可靠诊断工具的需求。ONCOREUM评分旨在区分表现为关节病的恶性肿瘤与JIA,并在初步验证中表现出高性能。然而,其效用仅限于患有关节病的患者,并不适用于其他形式的MSK受累。本研究旨在验证ONCOREUM评分在关节病患者中的有效性,并开发一个扩展的预测模型,以区分癌症相关的MSK症状与JIA。
纳入年龄小于16岁、诊断为癌症或JIA的患者。这项回顾性研究分两个阶段进行:(1)评估ONCOREUM评分识别伴有关节病的癌症的能力,(2)使用逐步逻辑回归分析开发一个模型,以区分癌症相关的MSK症状与JIA。
共审查了1026例患者(646例癌症患者,380例JIA患者)。在第1阶段,纳入了26例癌症患者(4.0%)和351例JIA患者(92.4%)。ONCOREUM评分(临界值=-6)的敏感性为88.5%,特异性为65.0%,曲线下面积(AUC)为0.77。在第2阶段,84例(13%)癌症病例出现MSK症状(61例血液系统疾病,23例实体瘤)。通过多变量分析获得了最佳拟合模型:背痛(比值比[OR]15.58,95%置信区间[CI]2.77-87.64,p<0.02)、夜间疼痛(OR 789.97,95%CI 51.26-12175.54,p<0.0001)、四肢骨疼痛(OR 24.11,95%CI 6.91-84.12,p<0.0001)、面色苍白(OR 6.30,95%CI 1.55-25.60,p<0.01)、晨僵(OR 0.03,95%CI 0.002-0.57,p<0.02)、附加性关节炎(OR 0.003,95%CI 0.00-0.04,p<0.0001)和单关节受累(OR 0.02,95%CI 0.00-0.23,p<0.00