Department of Neurosurgery, University Hospital Leipzig, 04103, Leipzig, Germany.
Department of Radiation Oncology, University Hospital Leipzig, 04103, Leipzig, Germany.
Acta Neuropathol Commun. 2023 Nov 28;11(1):189. doi: 10.1186/s40478-023-01690-y.
Homozygous CDKN2A/B deletion has been associated with an increased risk of recurrence in meningiomas. However, the evidence is confined to a limited number of studies, and the importance of heterozygous CDKN2A/B deletions remains insufficiently investigated. Hence, the present meta-analysis reconstructs individual patient data (IPD) and reconstructs the probabilities of progression-free survival (PFS) stratified by CDKN2A/B status. IPD of PFS rates were extracted from published Kaplan-Meier plots using the R package IPDfromKM in R studio (RStudio, Boston, MA, USA). Reconstructed Kaplan-Meier Plots of the pooled IPD data were created. One-stage and two-stage meta-analyses were performed. Hazard ratios (HR) were used as effective measures. Of 181 records screened, four articles with 2521 participants were included. The prevalence of homozygous CDKN2A/B deletions in the included studies was 0.049 (95% CI 0.040-0.057), with higher tumor grades associated with a significantly greater proportion of CDKN2A/B deletions. The reconstructed PFS curves for the pooled cohort showed that the median PFS time of patients with a CDKN2A/B wild-type status, heterozygous or homozygous CDKN2A/B deletion was 180.0 (95% CI 145.7-214.3), 26.1 (95% CI 23.3-29.0), and 11.00 (95% CI 8.6-13.3) months, respectively (p < 0.0001). Both hetero- or homozygous CDKN2A/B deletions were significantly associated with shortened time to meningioma progression. One-stage meta-analysis showed that hetero- (HR: 5.5, 95% CI 4.0-7.6, p < 0.00001) and homozygous CDKN2A/B deletions (HR: 8.4, 95% CI 6.4-11.0, p < 0.00001) are significantly associated with shortened time to meningioma progression. Multivariable Cox regression analysis of progression in a subgroup with available covariates (age, sex, WHO grade, and TERT status) and also two-stage meta-analysis confirmed and validated the results of the one-stage analysis that both heterozygous and homozygous CDKN2A/B deletions are of prognostic importance. Further large-scale studies of WHO grade 2 and 3 meningiomas are needed to validate the importance of heterozygous CDKN2A/B deletions with consideration of established factors.
纯合性 CDKN2A/B 缺失与脑膜瘤复发风险增加相关。然而,证据仅限于少数研究,杂合性 CDKN2A/B 缺失的重要性仍未得到充分研究。因此,本荟萃分析重建了个体患者数据(IPD),并按 CDKN2A/B 状态分层重建无进展生存率(PFS)的概率。从已发表的 Kaplan-Meier 图中使用 R 工作室中的 R 包 IPDfromKM(波士顿,MA,美国)提取 PFS 率的 IPD。创建了汇总 IPD 数据的重建 Kaplan-Meier 图。进行了单阶段和两阶段荟萃分析。风险比(HR)用作有效措施。在筛选出的 181 份记录中,纳入了 4 篇包含 2521 名参与者的文章。纳入研究中纯合性 CDKN2A/B 缺失的患病率为 0.049(95%CI 0.040-0.057),肿瘤分级较高与 CDKN2A/B 缺失的比例显著增加相关。汇总队列的重建 PFS 曲线表明,CDKN2A/B 野生型、杂合或纯合性 CDKN2A/B 缺失患者的中位 PFS 时间分别为 180.0(95%CI 145.7-214.3)、26.1(95%CI 23.3-29.0)和 11.00(95%CI 8.6-13.3)个月(p<0.0001)。杂合或纯合性 CDKN2A/B 缺失均与脑膜瘤进展时间缩短显著相关。单阶段荟萃分析显示,杂合性(HR:5.5,95%CI 4.0-7.6,p<0.00001)和纯合性 CDKN2A/B 缺失(HR:8.4,95%CI 6.4-11.0,p<0.00001)与脑膜瘤进展时间缩短显著相关。在具有可用协变量(年龄、性别、WHO 分级和 TERT 状态)的亚组中进行多变量 Cox 回归分析,以及两阶段荟萃分析均证实并验证了单阶段分析的结果,即杂合性和纯合性 CDKN2A/B 缺失均具有预后意义。需要进一步对 WHO 分级 2 级和 3 级脑膜瘤进行大规模研究,以考虑已确定的因素来验证杂合性 CDKN2A/B 缺失的重要性。