Overall survival and progression-free survival in pediatric meningiomas: a systematic review and individual patient-level meta-analysis.

IF 3.2 2区医学 Q2 CLINICAL NEUROLOGY

Journal of Neuro-Oncology Pub Date : 2025-01-09 DOI:10.1007/s11060-024-04917-7

Johannes Wach, Martin Vychopen, Alim Emre Basaran, Marcos Tatagiba, Roland Goldbrunner, Erdem Güresir

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引用次数: 0

Abstract

Background: Pediatric meningiomas (PMs) are rare central nervous system tumors, accounting for 1-5% of all meningiomas, and differ from adult meningiomas in clinical, histopathological, and molecular features. Current guidelines primarily focus on adults, leaving a gap in evidence-based management for PMs. This study presents the largest meta-analysis of longitudinal individual patient data (IPD) to date, addressing progression-free survival (PFS) and overall survival (OS) in pediatric patients.

Methods: Data from 20 studies (2011-2023), including 1010 pediatric meningioma cases, were analyzed to assess PFS and OS stratified by WHO grade, NF1/NF2 status, extent of resection (EOR), and adjuvant radiotherapy. Longitudinal survival data were reconstructed from Kaplan-Meier curves using IPD extraction methods.

Results: PMs affect males and females nearly equally (52.1% vs. 47.9%). WHO grade 3 tumors had significantly shorter PFS (72.1 months) compared to grades 1 (209.8 months) and 2 (137.5 months) (p < 0.001). No significant OS difference between WHO grades 1 and 2 PMs were observed. NF1- and NF2-associated tumors showed shorter PFS (59.7 and 138.4 months) than sporadic cases (180.6 months) (p = 0.02). GTR significantly improved PFS (113.8 vs. 40.1 months, p < 0.001) and OS (602.9 vs. 173.8 months, p < 0.001). Radiotherapy enhanced PFS (72.5 vs. 23.8 months, p = 0.009) and OS (140.7 vs. 63.0 months, p = 0.002) in grade 3 tumors but not in WHO grade 2 PMs (p = 0.43).

Conclusions: This largest meta-analysis highlights the critical roles of GTR and adjuvant radiotherapy in improving outcomes for high-grade PMs and underscores the urgent need for pediatric-specific management guidelines based on robust longitudinal data.

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儿童脑膜瘤的总生存期和无进展生存期：一项系统评价和个体患者水平的荟萃分析。

背景：小儿脑膜瘤是一种罕见的中枢神经系统肿瘤，占所有脑膜瘤的1-5%，在临床、组织病理学和分子特征上与成人脑膜瘤不同。目前的指导方针主要针对成年人，在pm的循证管理方面留下了空白。这项研究提出了迄今为止最大的纵向个体患者数据（IPD）荟萃分析，解决了儿科患者的无进展生存期（PFS）和总生存期（OS）。方法：分析20项研究（2011-2023年）的数据，包括1010例儿童脑膜瘤病例，以WHO分级、NF1/NF2状态、切除程度（EOR）和辅助放疗来评估PFS和OS。利用IPD提取方法从Kaplan-Meier曲线重构纵向生存数据。结果：经前综合症对男性和女性的影响几乎相等（52.1%对47.9%）。与1级（209.8个月）和2级（137.5个月）相比，WHO 3级肿瘤的PFS（72.1个月）显著缩短(p)。结论：这项最大的荟萃分析强调了GTR和辅助放疗在改善高级别PMs预后方面的关键作用，并强调了迫切需要基于可靠的纵向数据制定儿科特异性管理指南。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Journal of Neuro-Oncology 医学-临床神经学

CiteScore

6.60

自引率

7.70%

发文量

277

审稿时长

3.3 months

期刊介绍： The Journal of Neuro-Oncology is a multi-disciplinary journal encompassing basic, applied, and clinical investigations in all research areas as they relate to cancer and the central nervous system. It provides a single forum for communication among neurologists, neurosurgeons, radiotherapists, medical oncologists, neuropathologists, neurodiagnosticians, and laboratory-based oncologists conducting relevant research. The Journal of Neuro-Oncology does not seek to isolate the field, but rather to focus the efforts of many disciplines in one publication through a format which pulls together these diverse interests. More than any other field of oncology, cancer of the central nervous system requires multi-disciplinary approaches. To alleviate having to scan dozens of journals of cell biology, pathology, laboratory and clinical endeavours, JNO is a periodical in which current, high-quality, relevant research in all aspects of neuro-oncology may be found.