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立体定向放射外科治疗中枢神经系统血管母细胞瘤的系统评价与Meta分析

Stereotactic radiosurgery in the management of central nervous system hemangioblastomas: a systematic review and meta-analysis.

作者信息

Zare Amirhossein, Zare Amirhessam, Khaboushan Alireza Soltani, Hajikarimloo Bardia, Sheehan Jason P

机构信息

Department of Neurosurgery, Tehran University of Medical Sciences, Tehran, Iran.

Department of Neurological Surgery, University of Virginia, Charlottesville, VA, USA.

出版信息

Neurosurg Rev. 2025 Mar 17;48(1):303. doi: 10.1007/s10143-025-03454-9.

Abstract

Central nervous system (CNS) hemangioblastomas are rare, benign vascular tumors occurring sporadically or with von Hippel-Lindau (VHL) disease. While resection remains the primary treatment for symptomatic lesions, stereotactic radiosurgery (SRS) has emerged as an alternative where surgery is not feasible. This research aims to investigate the efficacy and safety of SRS for CNS hemangioblastomas, focusing on long-term outcomes and identifying key prognostic factors. A systematic search was conducted in PubMed, Scopus, Embase, Web of Science, and Cochrane Library till October 4th, 2024. Studies investigating the efficacy of SRS for CNS hemangioblastomas were included. The risk of bias was assessed using the ROBINS-I tool. Meta-analysis, subgroup analysis, and meta-regression were performed using the R programming language. A total of 28 studies with 627 patients and 1761 lesions were included. Our meta-analysis demonstrated pooled overall, 5- and 10-year local tumor control rates of 89% (95% CI: 85-92%), 87% (95% CI: 82-91%), and 80% (95% CI: 63-91%), respectively. Tumor response assessment revealed that 28% (95% CI: 19-40%) of lesions had regression, while 59% (95% CI: 46-70%) remained stable. VHL-associated lesions had a higher control than sporadic tumors at the 5-year follow-up (VHL: 94% (95% CI: 90-97%) vs. Sporadic: 82% (95% CI: 76-86%), P < 0.01). Pooled adverse events were 11% (95% CI: 8-15%). Meta-regression identified the female sex as a positive predictor for tumor control and lower adverse event rate (P < 0.01 and P = 0.02, respectively), while higher marginal and maximum radiation doses correlated with increased adverse events (P = 0.02 and P = 0.03, respectively). SRS represents a reasonably effective and safe treatment option for CNS hemangioblastomas, particularly in VHL-associated lesions. Patient demographics, VHL status, and tumor characteristics have been analyzed to identify factors potentially impacting treatment outcomes.

摘要

中枢神经系统(CNS)血管母细胞瘤是罕见的良性血管肿瘤,可散发出现或与冯·希佩尔-林道(VHL)病相关。虽然手术切除仍是有症状病变的主要治疗方法,但立体定向放射外科(SRS)已成为手术不可行时的替代方案。本研究旨在调查SRS治疗CNS血管母细胞瘤的疗效和安全性,重点关注长期结果并确定关键预后因素。截至2024年10月4日,在PubMed、Scopus、Embase、Web of Science和Cochrane图书馆进行了系统检索。纳入了研究SRS治疗CNS血管母细胞瘤疗效的研究。使用ROBINS-I工具评估偏倚风险。使用R编程语言进行荟萃分析、亚组分析和元回归。共纳入28项研究,涉及627例患者和1761个病灶。我们的荟萃分析显示,汇总的总体、5年和10年局部肿瘤控制率分别为89%(95%CI:85-92%)、87%(95%CI:82-91%)和80%(95%CI:63-91%)。肿瘤反应评估显示,28%(95%CI:19-40%)的病灶有消退,而59%(95%CI:46-70%)保持稳定。在5年随访时,与VHL相关的病灶比散发性肿瘤有更高的控制率(VHL:94%(95%CI:90-97%)对散发性:82%(95%CI:76-86%),P<0.01)。汇总的不良事件发生率为11%(95%CI:8-15%)。元回归确定女性为肿瘤控制的阳性预测因素且不良事件发生率较低(分别为P<0.01和P=0.02),而较高的边缘和最大放射剂量与不良事件增加相关(分别为P=0.02和P=0.03)。SRS是治疗CNS血管母细胞瘤的一种相当有效且安全的治疗选择,尤其是在与VHL相关的病灶中。已分析患者人口统计学、VHL状态和肿瘤特征,以确定可能影响治疗结果的因素。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ee59/11911270/644e871b28a3/10143_2025_3454_Fig1_HTML.jpg

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