{"title":"鉴别癌症相关肌肉骨骼症状与青少年特发性关节炎的特征并建立预测模型。","authors":"Khwanjira Archawanantakul, Soamarat Vilaiyuk, Samart Pakakasama, Butsabong Lerkvaleekul","doi":"10.1186/s12969-025-01121-3","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>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.</p><p><strong>Methods: </strong>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.</p><p><strong>Results: </strong>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).</p><p><strong>Conclusions: </strong>The refined predictive model is a promising clinical tool for differentiating cancer-associated MSK symptoms from JIA.</p>","PeriodicalId":54630,"journal":{"name":"Pediatric Rheumatology","volume":"23 1","pages":"73"},"PeriodicalIF":2.3000,"publicationDate":"2025-07-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12261695/pdf/","citationCount":"0","resultStr":"{\"title\":\"Identifying the characteristics of and developing a predictive model for differentiating cancer-related musculoskeletal symptoms from juvenile idiopathic arthritis.\",\"authors\":\"Khwanjira Archawanantakul, Soamarat Vilaiyuk, Samart Pakakasama, Butsabong Lerkvaleekul\",\"doi\":\"10.1186/s12969-025-01121-3\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Background: </strong>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.</p><p><strong>Methods: </strong>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.</p><p><strong>Results: </strong>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). 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引用次数: 0
摘要
背景:肌肉骨骼(MSK)症状是儿科患者的常见症状,可能由一系列疾病引起,包括幼年特发性关节炎(JIA)和恶性肿瘤。由于重叠的临床特征,在最初的表现中区分癌症相关的MSK症状和JIA仍然具有挑战性。恶性肿瘤诊断的延误可导致显著的发病率,强调需要可靠的诊断工具。ONCOREUM评分用于区分以关节病变为表现的恶性肿瘤和JIA,并在初步验证中表现出高性能。然而,它的效用仅限于关节病患者,而不能扩展到其他形式的MSK受损伤。本研究旨在验证ONCOREUM评分在关节病患者中的应用,并建立一个扩展的预测模型,以区分癌症相关的MSK症状和JIA。结果:共纳入1026例患者(肿瘤646例,JIA 380例)。一期共纳入26例肿瘤患者(4.0%)和351例JIA患者(92.4%)。ONCOREUM评分(截止值= - 6)的敏感性为88.5%,特异性为65.0%,AUC为0.77。在第二阶段,84例(13%)癌症患者出现MSK症状(61例血液病,23例实体瘤)。通过多变量分析获得最佳拟合模型:背部疼痛(OR 15.58, 95% CI 2.77-87.64, p)。结论:改进的预测模型是鉴别癌症相关MSK症状与JIA的一种有前景的临床工具。
Identifying the characteristics of and developing a predictive model for differentiating cancer-related musculoskeletal symptoms from juvenile idiopathic arthritis.
Background: 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.
Methods: 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.
Results: 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).
Conclusions: The refined predictive model is a promising clinical tool for differentiating cancer-associated MSK symptoms from JIA.
期刊介绍:
Pediatric Rheumatology is an open access, peer-reviewed, online journal encompassing all aspects of clinical and basic research related to pediatric rheumatology and allied subjects.
The journal’s scope of diseases and syndromes include musculoskeletal pain syndromes, rheumatic fever and post-streptococcal syndromes, juvenile idiopathic arthritis, systemic lupus erythematosus, juvenile dermatomyositis, local and systemic scleroderma, Kawasaki disease, Henoch-Schonlein purpura and other vasculitides, sarcoidosis, inherited musculoskeletal syndromes, autoinflammatory syndromes, and others.