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地诺单抗时代之前骨转移性巨细胞瘤的预后。一项系统评价和荟萃分析。

Prognosis of metastatic giant cell tumor of bone in the pre-denosumab era. A systematic review and a meta-analysis.

作者信息

Itkin Boris, Straminsky Samanta, De Ronato Gabriela, Lewi Daniel, Marantz Adolfo, Bardach Ariel

机构信息

Department of Medical Oncology, Juan A. Fernandez Hospital.

Institute for Clinical Effectiveness and Health Policy, Center for Research in Epidemiology and Public Health, National Scientific and Technical Research Council, Argentina.

出版信息

Jpn J Clin Oncol. 2018 Jul 1;48(7):640-652. doi: 10.1093/jjco/hyy067.

DOI:10.1093/jjco/hyy067
PMID:29741702
Abstract

BACKGROUND

Data on long-term prognosis of metastatic GCT (mGCT) is scant. The frequency of spontaneous regressions (SRs) is unknown. We aimed to estimate the prognosis of mGCT.

METHODS

We searched electronic scientific literature databases and generic Internet from January 1980 to August 2017. After identifying eligible studies we performed descriptive analyses and meta-analyses to estimate overall survival (OS), disease specific survival (DSS) and frequency of SRs in the years before the widespread use of denosumab. We performed pre-specified subgroup analyses of studies published before and after 2000 and of those with more and less than 10 years of follow-up.

RESULTS

After retrieving and combining data from 26 relevant retrospective case-series totaling 242 patients with a median follow-up of 6.9 years, the estimated pooled OS was 86.9% (95% CI 78.0-94.2). Pooled DSS was 88.0% (95% CI 79.7-94.7). SRs were observed in 4.5% of patients. In the subgroup of studies published after 2000 mGCT was the only cause of death of affected subjects. In case-series with a follow-up longer than 10 years pooled DSS was 69.7% (95% CI 25.5-99.8).

CONCLUSIONS

To our knowledge this is the first study to derive estimated pooled OS and DSS of mGCT based on a large dataset. SRs were not exceptional phenomena. In a long run the disease could impact in a significant way on the life expectancy of affected subjects.

摘要

背景

转移性生殖细胞肿瘤(mGCT)的长期预后数据稀少。自发消退(SR)的频率尚不清楚。我们旨在评估mGCT的预后。

方法

我们检索了1980年1月至2017年8月的电子科学文献数据库和普通互联网。在确定符合条件的研究后,我们进行了描述性分析和荟萃分析,以估计在地诺单抗广泛使用之前数年的总生存期(OS)、疾病特异性生存期(DSS)和SR频率。我们对2000年之前和之后发表的研究以及随访时间超过和少于10年的研究进行了预先指定的亚组分析。

结果

从26个相关回顾性病例系列中检索并合并数据,共242例患者,中位随访时间为6.9年,估计汇总OS为86.9%(95%CI 78.0-94.2)。汇总DSS为88.0%(95%CI 79.7-94.7)。4.5%的患者出现SR。在2000年之后发表的研究亚组中,mGCT是受影响受试者的唯一死亡原因。在随访时间超过10年的病例系列中,汇总DSS为69.7%(95%CI 25.5-99.8)。

结论

据我们所知,这是第一项基于大型数据集得出mGCT估计汇总OS和DSS的研究。SR并非罕见现象。从长远来看,该疾病可能会对受影响受试者的预期寿命产生重大影响。

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