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Survival after resection of malignant peripheral nerve sheath tumors: Introducing and validating a novel type-specific prognostic model.
Acem, Ibtissam; Steyerberg, Ewout W; Spreafico, Marta; Grünhagen, Dirk J; Callegaro, Dario; Spinner, Robert J; Pendleton, Courtney; Coert, J Henk; Miceli, Rosalba; Abruzzese, Giulia; Flucke, Uta E; Slooff, Willem-Bart M; van Dalen, Thijs; Been, Lukas B; Bonenkamp, Han J; Anten, Monique H M E; Broen, Martinus P G; Bemelmans, Marc H A; Bramer, Jos A M; Schaap, Gerard R; Kievit, Arthur J; van der Hage, Jos; van Houdt, Winan J; van de Sande, Michiel A J; Gronchi, Alessandro; Verhoef, Cornelis; Martin, Enrico.
Afiliación
  • Acem I; Department of Surgical Oncology and Gastrointestinal Surgery,Erasmus MC Cancer Institute, >Rotterdam, The Netherlands.
  • Steyerberg EW; Department of Orthopedic Oncology, Leiden University Medical Centre, >Leiden, The Netherlands.
  • Spreafico M; Department of Biomedical Data Sciences, Leiden University Medical Centre, >Leiden, The Netherlands.
  • Grünhagen DJ; Department of Medical Statistics, Mathematical Institute, Leiden University, >Leiden, The Netherlands.
  • Callegaro D; Department of Surgical Oncology and Gastrointestinal Surgery,Erasmus MC Cancer Institute, >Rotterdam, The Netherlands.
  • Spinner RJ; Department of Surgery, Fondazione IRCCS Istituto Nazionale dei Tumori, >Milan, Italy.
  • Pendleton C; Department of Neurosurgery, Mayo Clinic, >Rochester, Minnesota, USA.
  • Coert JH; Department of Neurosurgery, Stony Brook University School of Medicine, Stony Brook, New York, USA.
  • Miceli R; Department of Reconstructive Surgery, University Medical Centre Utrecht, >Utrecht, The Netherlands.
  • Abruzzese G; Department of Clinical Epidemiology and Trial Organization, Fondazione IRCCS Istituto Nazionale dei Tumori, >Milan, Italy.
  • Flucke UE; Department of Surgery, Fondazione IRCCS Istituto Nazionale dei Tumori, >Milan, Italy.
  • Slooff WM; Department of Pathology, Radboud University Medical Centre, >Nijmegen, The Netherlands.
  • van Dalen T; Department of Neurosurgery, University Medical Centre Utrecht, >Utrecht, The Netherlands.
  • Been LB; Department of Surgical Oncology and Gastrointestinal Surgery,Erasmus MC Cancer Institute, >Rotterdam, The Netherlands.
  • Bonenkamp HJ; Department of Surgical Oncology, University Medical Centre Groningen, Groningen, The Netherlands.
  • Anten MHME; Department of Surgical Oncology, Radboud University Medical Centre, >Nijmegen, The Netherlands.
  • Broen MPG; Department of Neurology, Maastricht University Medical Centre, >Maastricht, The Netherlands.
  • Bemelmans MHA; Department of Neurology, Maastricht University Medical Centre, >Maastricht, The Netherlands.
  • Bramer JAM; Department of Surgical Oncology, Maastricht University Medical Centre, >Maastricht, The Netherlands.
  • Schaap GR; Department of Orthopedic Surgery, Amsterdam University Medical Centre, Amsterdam, The Netherlands.
  • Kievit AJ; Department of Orthopedic Surgery, Amsterdam University Medical Centre, Amsterdam, The Netherlands.
  • van der Hage J; Department of Orthopedic Surgery, Amsterdam University Medical Centre, Amsterdam, The Netherlands.
  • van Houdt WJ; Department of Surgical Oncology, Leiden University Medical Centre, Leiden, The Netherlands.
  • van de Sande MAJ; Department of Surgical Oncology, The Netherlands Cancer Institute, Amsterdam, The Netherlands.
  • Gronchi A; Department of Orthopedic Oncology, Leiden University Medical Centre, >Leiden, The Netherlands.
  • Verhoef C; Department of Surgery, Fondazione IRCCS Istituto Nazionale dei Tumori, >Milan, Italy.
  • Martin E; Department of Surgical Oncology and Gastrointestinal Surgery,Erasmus MC Cancer Institute, >Rotterdam, The Netherlands.
Neurooncol Adv ; 6(1): vdae083, 2024.
Article en En | MEDLINE | ID: mdl-38946881
ABSTRACT

Background:

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).

Methods:

This is a retrospective multicenter cohort study of patients with MPNST from 11 secondary or tertiary centers in The Netherlands, Italy and the United States of America. All patients diagnosed with primary MPNST who underwent macroscopically complete surgical resection from 2000 to 2019 were included in this study. A multivariable Cox proportional hazard model for OS was estimated with prespecified 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.

Results:

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.

Conclusions:

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.
Palabras clave

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: Neurooncol Adv Año: 2024 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: Neurooncol Adv Año: 2024 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Reino Unido