Survival after Resection of Malignant Peripheral Nerve Sheath Tumors: introducing and validating a novel type-specific prognostic model

IF 4.3 3区材料科学 Q1 ENGINEERING, ELECTRICAL & ELECTRONIC

ACS Applied Electronic Materials Pub Date : 2024-06-05 DOI:10.1093/noajnl/vdae083

I. Acem, E. Steyerberg, Marta Spreafico, D. Grünhagen, D. Callegaro, Robert J Spinner, Courtney Pendleton, J. H. Coert, Rosalba Miceli, Giulia Abruzzese, U. Flucke, Willem-Bart M. Slooff, T. van Dalen, L. B. Been, H. Bonenkamp, M. Anten, Martinus P G Broen, Marc H A Bemelmans, J. Bramer, Gerard Schaap, A. Kievit, J. van der Hage, W. V. van Houdt, M. V. D. van de Sande, A. Gronchi, C. Verhoef, E. Martin

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引用次数: 0

Abstract

This study aimed to assess the performance of currently available risk calculators in a cohort of patients with malignant peripheral nerve sheath tumors (MPNST) and to create an MPNST-specific prognostic model including type-specific predictors for overall survival (OS). This is a retrospective multicenter cohort study of patients with MPNST from eleven secondary or tertiary centers in The Netherlands, Italy and the USA. All patients diagnosed with primary MPNST who underwent macroscopically complete surgical resection from 2000-2019 were included in this study. A multivariable Cox proportional hazard model for OS was estimated with pre-specified predictors (age, grade, size, NF-1 status, triton status, depth, tumor location and surgical margin). Model performance was assessed for the Sarculator and PERSARC calculators by examining discrimination (C-index) and calibration (calibration plots and observed-expected statistic; O/E-statistic). Internal-external cross-validation by different regions was performed to evaluate the generalizability of the model. A total of 507 patients with primary MPNSTs were included from 11 centers in 7 regions. During follow-up (median 8.7 years), 211 patients died. The C-index was 0.60 (95% CI 0.53-0.67) for both Sarculator and PERSARC. The MPNST-specific model had a pooled C-index of 0.69 (95%CI 0.65-0.73) at validation, with adequate discrimination and calibration across regions. The MPNST-specific MONACO model can be used to predict 3-, 5-, and 10-year OS in patients with primary MPNST who underwent macroscopically complete surgical resection. Further validation may refine the model to inform patients and physicians on prognosis and support them in shared decision-making.

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恶性周围神经鞘瘤切除术后的存活率：引入并验证新型特异性预后模型

这项研究旨在评估目前可用的风险计算器在恶性周围神经鞘瘤（MPNST）患者队列中的表现，并建立一个MPNST特异性预后模型，包括总生存期（OS）的特异性预测因子。这是一项回顾性多中心队列研究，研究对象是来自荷兰、意大利和美国 11 个二级或三级中心的 MPNST 患者。本研究纳入了2000年至2019年期间所有被诊断为原发性骨髓增生性肿瘤并接受了宏观完整手术切除的患者。利用预先指定的预测因子（年龄、分级、大小、NF-1状态、triton状态、深度、肿瘤位置和手术切缘）估算了OS的多变量Cox比例危险模型。通过检查区分度（C-指数）和校准（校准图和观察-预期统计量；O/E-统计量）来评估 Sarculator 和 PERSARC 计算器的模型性能。为评估模型的通用性，还按不同地区进行了内部和外部交叉验证。共纳入了来自 7 个地区 11 个中心的 507 名原发性 MPNST 患者。在随访期间（中位 8.7 年），211 名患者死亡。Sarculator 和 PERSARC 的 C 指数均为 0.60（95% CI 0.53-0.67）。MPNST特异性模型在验证时的汇总C指数为0.69（95%CI 0.65-0.73），在不同地区具有足够的区分度和校准性。 MPNST特异性MONACO模型可用于预测接受宏观完整手术切除的原发性MPNST患者的3年、5年和10年OS。进一步的验证可完善该模型，为患者和医生提供预后信息，支持他们共同决策。

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来源期刊

ACS Applied Electronic Materials Multiple-

CiteScore

7.20

自引率

4.30%

发文量

567

期刊介绍： ACS Applied Electronic Materials is an interdisciplinary journal publishing original research covering all aspects of electronic materials. The journal is devoted to reports of new and original experimental and theoretical research of an applied nature that integrate knowledge in the areas of materials science, engineering, optics, physics, and chemistry into important applications of electronic materials. Sample research topics that span the journal's scope are inorganic, organic, ionic and polymeric materials with properties that include conducting, semiconducting, superconducting, insulating, dielectric, magnetic, optoelectronic, piezoelectric, ferroelectric and thermoelectric. Indexed/Abstracted： Web of Science SCIE Scopus CAS INSPEC Portico