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Efficacy of Immune Checkpoint Inhibitors in Rare Tumours: A Systematic Review
Frontiers in Immunology ( IF 7.3 ) Pub Date : 2021-09-20 , DOI: 10.3389/fimmu.2021.720748
Fausto Petrelli 1 , Francesca Consoli 2 , Antonio Ghidini 3 , Gianluca Perego 4 , Andrea Luciani 1 , Paola Mercurio 5 , Alfredo Berruti 2 , Salvatore Grisanti 2
Affiliation  

Background

Rare cancers, as defined by the European Union, occur in fewer than 15 out of 100,000 people each year. The International Rare Cancer Consortium defines rare cancer incidence as less than six per 100,000 per year. There is a growing number of reports of the efficacy of immune checkpoint inhibitor (ICI) therapy in patients with rare tumours, and hence, we conducted a comprehensive review to summarise and analyse the available literature.

Methods

A literature search of PubMed was performed on January 31, 2021, using the following ICI names as keywords: ipilimumab, tremelimumab, cemiplimab, nivolumab, pembrolizumab, avelumab, atezolizumab, and durvalumab. Studies on patients with rare tumours who were being treated with ICIs were included. We plotted the overall response rate against the corresponding median survival across a variety of cancer types using linear regression.

Results

From 1,255 publications retrieved during the primary search, 62 publications were selected (with a total of 4,620 patients). Only four were randomised trials. A minority were first-line studies, while the remaining were studies in which ICIs were delivered as salvage therapy in pretreated patients. There was a good correlation between response rate and overall survival (Spearman R2 >0.9) in skin cancers, mesothelioma, and sarcomas.

Conclusions

Treatment of advanced-stage rare tumours with ICI therapy was found to be associated with significant activity in some orphan diseases (e.g., Merkel cell carcinoma) and hepatocellular carcinoma. Several ongoing prospective clinical trials will expand the knowledge on the safety and efficacy of ICI therapy in patients with these rare cancers.



中文翻译:

免疫检查点抑制剂在罕见肿瘤中的疗效:系统评价

Background

根据欧盟的定义,每年 100,000 人中不到 15 人患罕见癌症。国际罕见癌症联盟将罕见癌症的发病率定义为每年每 100,000 人中少于 6 人。越来越多的关于免疫检查点抑制剂(ICI)治疗罕见肿瘤患者疗效的报告,因此,我们进行了全面审查,以总结和分析现有文献。

Methods

2021 年 1 月 31 日对 PubMed 进行了文献检索,使用以下 ICI 名称作为关键词:ipilimumab、tremelimumab、cemiplimab、nivolumab、pembrolizumab、avelumab、atezolizumab 和 durvalumab。对接受 ICI 治疗的罕见肿瘤患者的研究也包括在内。我们使用线性回归绘制了各种癌症类型的总体反应率与相应的中位生存率。

Results

从初步检索期间检索到的 1,255 份出版物中,选择了 62 份出版物(总共 4,620 名患者)。只有四个是随机试验。少数是一线研究,而其余的研究是将 ICI 作为预先治疗过的患者的抢救疗法。在皮肤癌、间皮瘤和肉瘤中,反应率和总生存期(Spearman R 2 > 0.9)之间存在良好的相关性。

Conclusions

发现用 ICI 疗法治疗晚期罕见肿瘤与某些孤儿疾病(例如默克尔细胞癌)和肝细胞癌的显着活性有关。几项正在进行的前瞻性临床试验将扩大有关 ICI 治疗在这些罕见癌症患者中的安全性和有效性的知识。

更新日期:2021-09-20
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