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Background <p>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.</p> Methods <p>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.</p> Results <p>PMs affect males and females nearly equally (52.1% vs. 47.9%). WHO grade 3 tumors had significantly shorter PFS (72.1&#xa0;months) compared to grades 1 (209.8&#xa0;months) and 2 (137.5&#xa0;months) (<i>p</i> &lt; 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&#xa0;months) than sporadic cases (180.6&#xa0;months) (<i>p</i> = 0.02). GTR significantly improved PFS (113.8 vs. 40.1&#xa0;months, <i>p</i> &lt; 0.001) and OS (602.9 vs. 173.8&#xa0;months, <i>p</i> &lt; 0.001). Radiotherapy enhanced PFS (72.5 vs. 23.8&#xa0;months, <i>p</i> = 0.009) and OS (140.7 vs. 63.0&#xa0;months, <i>p</i> = 0.002) in grade 3 tumors but not in WHO grade 2 PMs (<i>p</i> = 0.43).</p> Conclusions <p>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.</p> Graphical Abstract <p></p>

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

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2025年1月9日

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

摘要

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.

Graphical Abstract