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Abstract：
BACKGROUND: Skeletal-related events (SREs), including the pathological fracture, surgical treatment or radiation of bone lesions, malignant spinal cord compression, hypercalcemia, are important considerations when managing metastatic bone tumors; however, owing to their rarity, the incidence of SREs in patients with Ewing sarcoma remains unknown.
METHODS: We retrospectively reviewed the clinical data from 146 patients with Ewing sarcoma treated at a single institution from 2005 to 2019. The median age at diagnosis was 22.7 years. Fifty patients (34.2%) had metastatic disease at diagnosis. The primary outcome was the SRE-free rate among patients with Ewing sarcoma. Moreover, we identified the risk factors for SREs using univariate or multivariate analyses.
RESULTS: During the observational period (median, 2.6 years), SREs occurred in 23 patients. Radiation to the bone, malignant spinal cord compression, and hypercalcemia were documented as the initial SREs in 12 patients (52.2%), 10 patients (43.5%), and one patient (4.3%), respectively. The SRE-free rate was 94.2 ± 2.0, 87.3 ± 3.0, and 79.6 ± 3.8% at 1, 2, and 3 years after the initial visit, respectively. Multivariate analysis revealed bone metastasis at diagnosis (hazard ratio [HR] = 4.41, p = 0.007), bone marrow invasion (HR = 34.08, p < 0.001), and local progression or recurrence after definitive treatment (HR = 3.98, p = 0.012) as independent risk factors for SREs.
CONCLUSIONS: SREs are non-rare events that can occur during the treatment course for Ewing sarcoma, with an especially high incidence of malignant spinal cord compression. Patients with metastatic disease at diagnosis, especially in the bone or bone marrow, or with local progression or recurrence after definitive treatment, should be carefully monitored for the occurrence of SREs. The most effective methods to monitor the occurrence of SREs and new preventative therapies for SREs should be investigated in the future.
METHODS: We retrospectively reviewed the clinical data from 146 patients with Ewing sarcoma treated at a single institution from 2005 to 2019. The median age at diagnosis was 22.7 years. Fifty patients (34.2%) had metastatic disease at diagnosis. The primary outcome was the SRE-free rate among patients with Ewing sarcoma. Moreover, we identified the risk factors for SREs using univariate or multivariate analyses.
RESULTS: During the observational period (median, 2.6 years), SREs occurred in 23 patients. Radiation to the bone, malignant spinal cord compression, and hypercalcemia were documented as the initial SREs in 12 patients (52.2%), 10 patients (43.5%), and one patient (4.3%), respectively. The SRE-free rate was 94.2 ± 2.0, 87.3 ± 3.0, and 79.6 ± 3.8% at 1, 2, and 3 years after the initial visit, respectively. Multivariate analysis revealed bone metastasis at diagnosis (hazard ratio [HR] = 4.41, p = 0.007), bone marrow invasion (HR = 34.08, p < 0.001), and local progression or recurrence after definitive treatment (HR = 3.98, p = 0.012) as independent risk factors for SREs.
CONCLUSIONS: SREs are non-rare events that can occur during the treatment course for Ewing sarcoma, with an especially high incidence of malignant spinal cord compression. Patients with metastatic disease at diagnosis, especially in the bone or bone marrow, or with local progression or recurrence after definitive treatment, should be carefully monitored for the occurrence of SREs. The most effective methods to monitor the occurrence of SREs and new preventative therapies for SREs should be investigated in the future.
摘要：
背景：骨骼相关事件（SRE），包括病理性骨折,骨损伤的手术治疗或放射，恶性脊髓压迫，高钙血症,是治疗转移性骨肿瘤时的重要考虑因素；然而，由于它们的稀有性,尤因肉瘤患者SREs的发生率尚不清楚.
方法：我们回顾性分析了2005年至2019年在单一机构治疗的146例尤文肉瘤患者的临床资料。诊断时的中位年龄为22.7岁。50名患者（34.2％）在诊断时患有转移性疾病。主要结果是Ewing肉瘤患者的无SRE率。此外，我们使用单因素或多因素分析确定了SRE的危险因素.
结果：在观察期间(中位数，2.6年),23例患者发生SREs。辐射到骨头，恶性脊髓压迫，和高钙血症被记录为12例患者的初始SRE（52.2％），10例（43.5%），和一名患者(4.3%)，分别。初次就诊后1年、2年和3年无SRE率为94.2±2.0、87.3±3.0和79.6±3.8%，分别。多因素分析显示诊断时骨转移（风险比[HR]=4.41，p=0.007），骨髓浸润（HR=34.08，p<0.001），最终治疗后局部进展或复发（HR=3.98，p=0.012）是SRE的独立危险因素。
结论：SREs是在尤文肉瘤治疗过程中可能发生的非罕见事件，恶性脊髓压迫的发生率尤其高。诊断时患有转移性疾病的患者，尤其是在骨骼或骨髓中，或局部进展或明确治疗后复发，应仔细监测SREs的发生。未来应研究监测SRE发生的最有效方法和新的SRE预防性治疗方法。
方法：我们回顾性分析了2005年至2019年在单一机构治疗的146例尤文肉瘤患者的临床资料。诊断时的中位年龄为22.7岁。50名患者（34.2％）在诊断时患有转移性疾病。主要结果是Ewing肉瘤患者的无SRE率。此外，我们使用单因素或多因素分析确定了SRE的危险因素.
结果：在观察期间(中位数，2.6年),23例患者发生SREs。辐射到骨头，恶性脊髓压迫，和高钙血症被记录为12例患者的初始SRE（52.2％），10例（43.5%），和一名患者(4.3%)，分别。初次就诊后1年、2年和3年无SRE率为94.2±2.0、87.3±3.0和79.6±3.8%，分别。多因素分析显示诊断时骨转移（风险比[HR]=4.41，p=0.007），骨髓浸润（HR=34.08，p<0.001），最终治疗后局部进展或复发（HR=3.98，p=0.012）是SRE的独立危险因素。
结论：SREs是在尤文肉瘤治疗过程中可能发生的非罕见事件，恶性脊髓压迫的发生率尤其高。诊断时患有转移性疾病的患者，尤其是在骨骼或骨髓中，或局部进展或明确治疗后复发，应仔细监测SREs的发生。未来应研究监测SRE发生的最有效方法和新的SRE预防性治疗方法。
