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

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 t...

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Vydané v:Journal of neuro-oncology Ročník 134; číslo 1; s. 231 - 240
Hlavní autori: Veldhuijzen van Zanten, Sophie E. M., Lane, Adam, Heymans, Martijn W., Baugh, Joshua, Chaney, Brooklyn, Hoffman, Lindsey M., Doughman, Renee, Jansen, Marc H. A., Sanchez, Esther, Vandertop, William P., Kaspers, Gertjan J. L., van Vuurden, Dannis G., Fouladi, Maryam, Jones, Blaise V., Leach, James
Médium: Journal Article
Jazyk:English
Vydavateľské údaje: New York Springer US 01.08.2017
Springer Nature B.V
Predmet:
Adolescent
Brain cancer
Brain Stem Neoplasms - diagnostic imaging
Brain Stem Neoplasms - mortality
Brain Stem Neoplasms - therapy
Brain tumors
Calibration
Chemotherapy
Child
Child, Preschool
Clinical Study
Cohort Studies
Female
Glioma
Glioma - diagnostic imaging
Glioma - mortality
Glioma - therapy
Humans
Image Processing, Computer-Assisted
Infant
Infant, Newborn
Kaplan-Meier Estimate
Magnetic Resonance Imaging
Male
Medicine
Medicine & Public Health
Neurology
Oncology
Prognosis
Registries
Regression Analysis
Risk groups
Survival
Cox proportional hazards modeling
Discrimination
Calibration
External validation
Prognostic modeling
ISSN:0167-594X, 1573-7373, 1573-7373
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Popis
Shrnutí: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.
Bibliografia:ObjectType-Article-1
SourceType-Scholarly Journals-1
ObjectType-Feature-2
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ISSN:0167-594X
1573-7373
1573-7373
DOI:10.1007/s11060-017-2514-9