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J Neurooncol. 2017 Aug;134(1):231-240. doi: 10.1007/s11060-017-2514-9. Epub 2017 May 30.

External validation of the diffuse intrinsic pontine glioma survival prediction model: a collaborative report from the International DIPG Registry and the SIOPE DIPG Registry.

Author information

1
Department of Paediatrics, Division of Oncology/Haematology, VU University Medical Center (VUmc), De Boelelaan 1118, 1081 HZ, Amsterdam, The Netherlands. s.veldhuijzen@vumc.nl.
2
Department of Paediatric Oncology/Haematology, VU University Medical Center, De Boelelaan 1118, Room KTC4.027, 1081 HZ, Amsterdam, The Netherlands. s.veldhuijzen@vumc.nl.
3
Department of Biostatistics and Epidemiology, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH, 45229, USA.
4
Department of Pediatrics, Cancer and Blood Diseases Institute, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH, 45229, USA.
5
Department of Epidemiology and Biostatistics, VU University Medical Center (VUmc), De Boelelaan 1089a, 1081 HV, Amsterdam, The Netherlands.
6
Department of Paediatrics, Division of Oncology/Haematology, VU University Medical Center (VUmc), De Boelelaan 1118, 1081 HZ, Amsterdam, The Netherlands.
7
Department of Radiology & Nuclear Medicine, VU University Medical Center (VUmc), De Boelelaan 1118, 1081 HZ, Amsterdam, The Netherlands.
8
Department of Neurosurgery, Neurosurgical Center Amsterdam, Room 2F 020, De Boelelaan 1117, 1081 HV, Amsterdam, The Netherlands.
9
Academy of Princess Máxima Center for Pediatric Oncology, Postbus 85090, 3508 AB, Utrecht, The Netherlands.
10
Department of Radiology, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH, 45229, USA.

Abstract

We aimed to perform external validation of the recently developed survival prediction model for diffuse intrinsic pontine glioma (DIPG), and discuss its utility. The DIPG survival prediction model was developed in a cohort of patients from the Netherlands, United Kingdom and Germany, registered in the SIOPE DIPG Registry, and includes age <3 years, longer symptom duration and receipt of chemotherapy as favorable predictors, and presence of ring-enhancement on MRI as unfavorable predictor. Model performance was evaluated by analyzing the discrimination and calibration abilities. External validation was performed using an unselected cohort from the International DIPG Registry, including patients from United States, Canada, Australia and New Zealand. Basic comparison with the results of the original study was performed using descriptive statistics, and univariate- and multivariable regression analyses in the validation cohort. External validation was assessed following a variety of analyses described previously. Baseline patient characteristics and results from the regression analyses were largely comparable. Kaplan-Meier curves of the validation cohort reproduced separated groups of standard (n = 39), intermediate (n = 125), and high-risk (n = 78) patients. This discriminative ability was confirmed by similar values for the hazard ratios across these risk groups. The calibration curve in the validation cohort showed a symmetric underestimation of the predicted survival probabilities. In this external validation study, we demonstrate that the DIPG survival prediction model has acceptable cross-cohort calibration and is able to discriminate patients with short, average, and increased survival. We discuss how this clinico-radiological model may serve a useful role in current clinical practice.

KEYWORDS:

Calibration; Cox proportional hazards modeling; Discrimination; External validation; Prognostic modeling

PMID:
28560664
PMCID:
PMC5543206
DOI:
10.1007/s11060-017-2514-9
[Indexed for MEDLINE]
Free PMC Article

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