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毛细血管扩张性骨肉瘤:与骨动脉瘤样囊肿鉴别的结果分析及诊断模型

Telangiectatic osteosarcoma: Outcome analyses and a diagnostic model for differentiation from aneurysmal bone cyst.

作者信息

Yin Jun-Qiang, Fu Yi-Wei, Xie Xian-Biao, Cheng Xiao-Yu, Yang Xiao-Yu, Liu Wei-Hai, Tu Jian, Gao Zhen-Hua, Shen Jing-Nan

机构信息

Department of Orthopedic Oncology, First Affiliated Hospital of Sun Yat-Sen University, 58 Second Zhongshan Road, Guangzhou, Guangdong, China.

Zhongshan School of Medicine, Sun Yat-Sen University, 74 Second Zhongshan Road, Guangzhou, Guangdong, China.

出版信息

J Bone Oncol. 2017 Nov 26;11:10-16. doi: 10.1016/j.jbo.2017.11.003. eCollection 2018 Jun.

Abstract

BACKGROUND AND PURPOSE

Telangiectatic osteosarcoma (TOS), a rare variant of osteosarcoma, may be easily misdiagnosed as aneurysmal bone cyst (ABC). The aims of this study were to investigate the diagnostic and prognostic factors of TOS by reviewing our experience with TOS and to develop a diagnostic model that may distinguish TOS from ABC.

MATERIALS AND METHODS

We identified 51 cases of TOS treated at the First Affiliated Hospital of Sun Yat-Sen University from March 2001 to January 2016 and reviewed their records, imaging information and pathological studies. A diagnostic model was developed to differentiate TOS and ABC by Bayes discriminant analysis and was evaluated. The log-rank test was used to analyze the prognostic factors of TOS and to compare the outcome differences between TOS and other high-grade osteosarcoma subtypes.

RESULTS

The multi-disciplinary diagnostic method employed that combined clinical, imaging, and pathological studies enhanced the diagnostic accuracy. Age 18 years or younger and pathologic fracture were more common among the TOS patients than among the ABC patients = .004 and .005, respectively). The average white blood cell (WBC), platelet, lactate dehydrogenase (LDH), and alkaline phosphatase (ALP) values of the TOS patients were higher than those of the ABC patients ( = .002, .003, .007, and .007, respectively). Our diagnostic model, including the aforementioned factors, accurately predicted 62% and 78% of the TOS patients in the training and validation sets, respectively. The 5-year estimates of event-free survival and overall survival of the TOS patients were 52.5 ± 9.4% and 54.9 ± 8.8%, respectively, which were similar to those of patients with other osteosarcoma subtypes ( = .950 and .615, respectively). Tumor volume and the LDH level were predictive prognostic factors ( = .040 and .044) but not the presence of pathologic fracture or misdiagnosis ( = .424 and .632, all respectively).

CONCLUSIONS

The multi-disciplinary diagnostic method and diagnostic model based on predictive factors, , age, the presence of pathologic fracture, and platelet, LDH, ALP and WBC levels, aided the differentiation of TOS and ABC. Smaller tumors and normal LDH levels were associated with better outcomes.

摘要

背景与目的

毛细血管扩张性骨肉瘤(TOS)是骨肉瘤的一种罕见变异型,容易被误诊为骨动脉瘤样囊肿(ABC)。本研究旨在通过回顾我们对TOS的诊治经验,探讨TOS的诊断和预后因素,并建立一个可将TOS与ABC区分开来的诊断模型。

材料与方法

我们确定了2001年3月至2016年1月在中山大学附属第一医院接受治疗的51例TOS患者,并回顾了他们的病历、影像资料和病理研究。通过贝叶斯判别分析建立了一个区分TOS和ABC的诊断模型并进行评估。采用对数秩检验分析TOS的预后因素,并比较TOS与其他高级别骨肉瘤亚型之间的预后差异。

结果

采用临床、影像和病理研究相结合的多学科诊断方法提高了诊断准确性。TOS患者中18岁及以下和病理性骨折比ABC患者更常见(分别为P = 0.004和0.005)。TOS患者的平均白细胞(WBC)、血小板、乳酸脱氢酶(LDH)和碱性磷酸酶(ALP)值高于ABC患者(分别为P = 0.002、0.003、0.007和0.007)。我们的诊断模型,包括上述因素,在训练集和验证集中分别准确预测了62%和78%的TOS患者。TOS患者的5年无事件生存率和总生存率估计分别为52.5±9.4%和54.9±8.8%,与其他骨肉瘤亚型患者相似(分别为P = 0.950和0.615)。肿瘤体积和LDH水平是预测预后的因素(P = 0.040和0.044),但病理性骨折或误诊的存在不是(分别为P = 0.424和0.632)。

结论

基于预测因素(年龄、病理性骨折的存在以及血小板、LDH、ALP和WBC水平)的多学科诊断方法和诊断模型有助于TOS与ABC的鉴别。较小的肿瘤和正常的LDH水平与较好的预后相关。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/27d3/5993956/4d51cb71ddba/gr1.jpg

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