Clinicopathological features and survival disparities between axial and appendicular skeletal Angiosarcoma: A SEER-based analysis using competing risk models

IF 3.5 2区医学 Q2 Medicine

Journal of Bone Oncology Pub Date : 2026-04-01 Epub Date: 2026-01-22 DOI:10.1016/j.jbo.2026.100740

Zhenliang Hao , Xuewei Zhang , Ning Yao , Kai Li , Luxin Lou , Xuan Zheng , Zhiming Guo , Jiabin Chen , Xuzhu Chen

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Abstract

Background

Primary angiosarcoma of bone (ASB) is a rare and aggressive primary bone tumor. The prognostic significance of anatomical location (axial vs. appendicular skeleton) remains poorly defined. This study aimed to compare clinicopathological features and survival outcomes between these two groups.

Methods

Patients diagnosed with primary ASB (1975–2019) were identified from the Surveillance, Epidemiology, and End Results (SEER) database. Tumors were categorized as axial (spine, pelvis, ribs, skull) or appendicular (extremities). Overall survival was analyzed using Kaplan-Meier and Cox models. Cancer-specific mortality (CSM) was evaluated with Fine-Gray competing risk regression to calculate subdistribution hazard ratios (SHR).

Results

Among 458 patients, 162 (35.4%) had axial and 296 (64.6%) had appendicular tumors. Axial patients were older (median 63 vs. 46 years, p < 0.001), had higher distant metastasis at diagnosis (43.2% vs. 29.1%, p = 0.002), and underwent surgery less frequently (37.0% vs. 81.1%, p < 0.001). The 5-year overall survival was significantly worse for axial tumors (16.8% vs. 38.2%, p < 0.001). The 5-year cumulative incidence of CSM was 69.5% for axial versus 50.8% for appendicular tumors (p < 0.001). Multivariate analysis confirmed axial location as an independent predictor of higher CSM (SHR 1.62, 95% CI: 1.25–2.10, p < 0.001) , independent of the year of diagnosis.

Conclusions

Axial tumors represent a distinct high-risk subgroup characterized by limited surgical resectability and inferior survival. Anatomical location should be considered a critical stratification factor in clinical management and future trials.

Abbreviations: ASB, Primary angiosarcoma of the bone; SEER, Surveillance, Epidemiology, and End Results; OS, Overall Survival; CSS, Cancer-Specific Survival; CSD, Cancer-Specific Death; CIF, Cumulative Incidence Function; SHR, Subdistribution Hazard Ratio; CI, Confidence Interval; HR, Hazard Ratio.

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轴状和尾状骨骼血管肉瘤的临床病理特征和生存差异：基于seer的竞争风险模型分析

原发性骨血管肉瘤（ASB）是一种罕见的侵袭性原发性骨肿瘤。解剖位置（轴骨与尾骨）的预后意义仍不明确。本研究旨在比较两组患者的临床病理特征和生存结果。方法从监测、流行病学和最终结果（SEER）数据库中确定1975-2019年诊断为原发性ASB的患者。肿瘤分为轴向肿瘤（脊柱、骨盆、肋骨、颅骨）和尾向肿瘤（四肢）。采用Kaplan-Meier和Cox模型分析总生存率。采用Fine-Gray竞争风险回归法评估癌症特异性死亡率（CSM），计算亚分布风险比（SHR）。结果458例患者中，轴端肿瘤162例（35.4%），尾端肿瘤296例（64.6%）。轴位患者年龄较大（中位为63岁对46岁，p < 0.001），诊断时远处转移率较高（43.2%对29.1%,p = 0.002），手术频率较低（37.0%对81.1%,p < 0.001）。轴状肿瘤的5年总生存率明显较差（16.8%比38.2%,p < 0.001）。轴向肿瘤5年累积发病率为69.5%，阑尾肿瘤为50.8% （p < 0.001）。多因素分析证实，轴位是CSM较高的独立预测因子（SHR 1.62, 95% CI: 1.25-2.10, p < 0.001），与诊断年份无关。结论输卵管肿瘤是一个独特的高危亚群，其特点是手术可切除性有限，生存期较低。在临床管理和未来的试验中，解剖位置应被视为一个关键的分层因素。缩写：ASB，原发性骨血管肉瘤；SEER、监测、流行病学和最终结果；OS：总生存期；CSS，癌症特异性生存；癌症特异性死亡；CIF，累积关联函数；亚分布风险比；CI，置信区间；HR，风险比。

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来源期刊

Journal of Bone Oncology ONCOLOGY-

CiteScore

7.20

自引率

2.90%

发文量

审稿时长

34 days

期刊介绍： The Journal of Bone Oncology is a peer-reviewed international journal aimed at presenting basic, translational and clinical high-quality research related to bone and cancer. As the first journal dedicated to cancer induced bone diseases, JBO welcomes original research articles, review articles, editorials and opinion pieces. Case reports will only be considered in exceptional circumstances and only when accompanied by a comprehensive review of the subject. The areas covered by the journal include: Bone metastases (pathophysiology, epidemiology, diagnostics, clinical features, prevention, treatment) Preclinical models of metastasis Bone microenvironment in cancer (stem cell, bone cell and cancer interactions) Bone targeted therapy (pharmacology, therapeutic targets, drug development, clinical trials, side-effects, outcome research, health economics) Cancer treatment induced bone loss (epidemiology, pathophysiology, prevention and management) Bone imaging (clinical and animal, skeletal interventional radiology) Bone biomarkers (clinical and translational applications) Radiotherapy and radio-isotopes Skeletal complications Bone pain (mechanisms and management) Orthopaedic cancer surgery Primary bone tumours Clinical guidelines Multidisciplinary care Keywords: bisphosphonate, bone, breast cancer, cancer, CTIBL, denosumab, metastasis, myeloma, osteoblast, osteoclast, osteooncology, osteo-oncology, prostate cancer, skeleton, tumour.