Survival prediction model of children with diffuse intrinsic pontine glioma based on clinical and radiological criteria

Marc H. Jansen; Sophie E. Veldhuijzen van Zanten; Esther Sanchez Aliaga; Martijn W. Heymans; Monika Warmuth-Metz; Darren Hargrave; Erica J. van der Hoeven; Corrie E. Gidding; Eveline S. de Bont; Omid S. Eshghi; Roel Reddingius; Cache M. Peeters; Antoinette Y. N. Schouten-van Meeteren; Rob H. J. Gooskens; Bernd Granzen; Gabriel M. Paardekooper; Geert O. Janssens; David P. Noske; Frederik Barkhof; Christof M. Kramm; W. Peter Vandertop; Gertjan J. Kaspers; Dennis G. van Vuurden

doi:10.1093/neuonc/nou104

Survival prediction model of children with diffuse intrinsic pontine glioma based on clinical and radiological criteria

Marc H. Jansen^*, Sophie E. Veldhuijzen van Zanten, Esther Sanchez Aliaga, Martijn W. Heymans, Monika Warmuth-Metz, Darren Hargrave, Erica J. van der Hoeven, Corrie E. Gidding, Eveline S. de Bont, Omid S. Eshghi, Roel Reddingius, Cache M. Peeters, Antoinette Y. N. Schouten-van Meeteren, Rob H. J. Gooskens, Bernd Granzen, Gabriel M. Paardekooper, Geert O. Janssens, David P. Noske, Frederik Barkhof, Christof M. KrammW. Peter Vandertop, Gertjan J. Kaspers, Dennis G. van Vuurden

^*Corresponding author for this work

Research output: Contribution to journal › Article › Academic › peer-review

123 Citations (Scopus)

Abstract

Background. Although diffuse intrinsic pontine glioma (DIPG) carries the worst prognosis of all pediatric brain tumors, studies on prognostic factors in DIPG are sparse. To control for confounding variables in DIPG studies, which generally include relatively small patient numbers, a survival prediction tool is needed.

Methods. A multicenter retrospective cohort study was performed in the Netherlands, the UK, and Germany with central review of clinical data and MRI scans of children with DIPG. Cox proportional hazards with backward regression was used to select prognostic variables (P<.05) to predict the accumulated 12-month risk of death. These predictors were transformed into a practical risk score. The model's performance was validated by bootstrapping techniques.

Results. A total of 316 patients were included. The median overall survival was 10 months. Multivariate Cox analysis yielded 5 prognostic variables of which the coefficients were included in the risk score. Age

Conclusions. We developed a DIPG survival prediction tool that can be used to predict the outcome of patients and for stratification in trials. Validation of the model is needed in a prospective cohort.

Original language	English
Pages (from-to)	160-166
Number of pages	7
Journal	Neuro-Oncology
Volume	17
Issue number	1
DOIs	https://doi.org/10.1093/neuonc/nou104
Publication status	Published - Jan-2015

Keywords

brainstem neoplasms
glioma
magnetic resonance imaging
pons
prognosis
BRAIN-STEM GLIOMA
CHILDHOOD
TUMORS
CHEMOTHERAPY
TRIALS

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Jansen, M. H., van Zanten, S. E. V., Aliaga, E. S., Heymans, M. W., Warmuth-Metz, M., Hargrave, D., van der Hoeven, E. J., Gidding, C. E., de Bont, E. S., Eshghi, O. S., Reddingius, R., Peeters, C. M., Schouten-van Meeteren, A. Y. N., Gooskens, R. H. J., Granzen, B., Paardekooper, G. M., Janssens, G. O., Noske, D. P., Barkhof, F., ... van Vuurden, D. G. (2015). Survival prediction model of children with diffuse intrinsic pontine glioma based on clinical and radiological criteria. Neuro-Oncology, 17(1), 160-166. https://doi.org/10.1093/neuonc/nou104

@article{48f6306097cd4b66bdfbd677d62ec1c6,

title = "Survival prediction model of children with diffuse intrinsic pontine glioma based on clinical and radiological criteria",

abstract = "Background. Although diffuse intrinsic pontine glioma (DIPG) carries the worst prognosis of all pediatric brain tumors, studies on prognostic factors in DIPG are sparse. To control for confounding variables in DIPG studies, which generally include relatively small patient numbers, a survival prediction tool is needed.Methods. A multicenter retrospective cohort study was performed in the Netherlands, the UK, and Germany with central review of clinical data and MRI scans of children with DIPG. Cox proportional hazards with backward regression was used to select prognostic variables (P<.05) to predict the accumulated 12-month risk of death. These predictors were transformed into a practical risk score. The model's performance was validated by bootstrapping techniques.Results. A total of 316 patients were included. The median overall survival was 10 months. Multivariate Cox analysis yielded 5 prognostic variables of which the coefficients were included in the risk score. AgeConclusions. We developed a DIPG survival prediction tool that can be used to predict the outcome of patients and for stratification in trials. Validation of the model is needed in a prospective cohort.",

keywords = "brainstem neoplasms, glioma, magnetic resonance imaging, pons, prognosis, BRAIN-STEM GLIOMA, CHILDHOOD, TUMORS, CHEMOTHERAPY, TRIALS",

author = "Jansen, {Marc H.} and {van Zanten}, {Sophie E. Veldhuijzen} and Aliaga, {Esther Sanchez} and Heymans, {Martijn W.} and Monika Warmuth-Metz and Darren Hargrave and {van der Hoeven}, {Erica J.} and Gidding, {Corrie E.} and {de Bont}, {Eveline S.} and Eshghi, {Omid S.} and Roel Reddingius and Peeters, {Cache M.} and {Schouten-van Meeteren}, {Antoinette Y. N.} and Gooskens, {Rob H. J.} and Bernd Granzen and Paardekooper, {Gabriel M.} and Janssens, {Geert O.} and Noske, {David P.} and Frederik Barkhof and Kramm, {Christof M.} and Vandertop, {W. Peter} and Kaspers, {Gertjan J.} and {van Vuurden}, {Dennis G.}",

year = "2015",

month = jan,

doi = "10.1093/neuonc/nou104",

language = "English",

volume = "17",

pages = "160--166",

journal = "Neuro-Oncology",

issn = "1522-8517",

publisher = "Oxford University Press",

number = "1",

}

Jansen, MH, van Zanten, SEV, Aliaga, ES, Heymans, MW, Warmuth-Metz, M, Hargrave, D, van der Hoeven, EJ, Gidding, CE, de Bont, ES, Eshghi, OS, Reddingius, R, Peeters, CM, Schouten-van Meeteren, AYN, Gooskens, RHJ, Granzen, B, Paardekooper, GM, Janssens, GO, Noske, DP, Barkhof, F, Kramm, CM, Vandertop, WP, Kaspers, GJ & van Vuurden, DG 2015, 'Survival prediction model of children with diffuse intrinsic pontine glioma based on clinical and radiological criteria', Neuro-Oncology, vol. 17, no. 1, pp. 160-166. https://doi.org/10.1093/neuonc/nou104

TY - JOUR

T1 - Survival prediction model of children with diffuse intrinsic pontine glioma based on clinical and radiological criteria

AU - Jansen, Marc H.

AU - van Zanten, Sophie E. Veldhuijzen

AU - Aliaga, Esther Sanchez

AU - Heymans, Martijn W.

AU - Warmuth-Metz, Monika

AU - Hargrave, Darren

AU - van der Hoeven, Erica J.

AU - Gidding, Corrie E.

AU - de Bont, Eveline S.

AU - Eshghi, Omid S.

AU - Reddingius, Roel

AU - Peeters, Cache M.

AU - Schouten-van Meeteren, Antoinette Y. N.

AU - Gooskens, Rob H. J.

AU - Granzen, Bernd

AU - Paardekooper, Gabriel M.

AU - Janssens, Geert O.

AU - Noske, David P.

AU - Barkhof, Frederik

AU - Kramm, Christof M.

AU - Vandertop, W. Peter

AU - Kaspers, Gertjan J.

AU - van Vuurden, Dennis G.

PY - 2015/1

Y1 - 2015/1

N2 - Background. Although diffuse intrinsic pontine glioma (DIPG) carries the worst prognosis of all pediatric brain tumors, studies on prognostic factors in DIPG are sparse. To control for confounding variables in DIPG studies, which generally include relatively small patient numbers, a survival prediction tool is needed.Methods. A multicenter retrospective cohort study was performed in the Netherlands, the UK, and Germany with central review of clinical data and MRI scans of children with DIPG. Cox proportional hazards with backward regression was used to select prognostic variables (P<.05) to predict the accumulated 12-month risk of death. These predictors were transformed into a practical risk score. The model's performance was validated by bootstrapping techniques.Results. A total of 316 patients were included. The median overall survival was 10 months. Multivariate Cox analysis yielded 5 prognostic variables of which the coefficients were included in the risk score. AgeConclusions. We developed a DIPG survival prediction tool that can be used to predict the outcome of patients and for stratification in trials. Validation of the model is needed in a prospective cohort.

AB - Background. Although diffuse intrinsic pontine glioma (DIPG) carries the worst prognosis of all pediatric brain tumors, studies on prognostic factors in DIPG are sparse. To control for confounding variables in DIPG studies, which generally include relatively small patient numbers, a survival prediction tool is needed.Methods. A multicenter retrospective cohort study was performed in the Netherlands, the UK, and Germany with central review of clinical data and MRI scans of children with DIPG. Cox proportional hazards with backward regression was used to select prognostic variables (P<.05) to predict the accumulated 12-month risk of death. These predictors were transformed into a practical risk score. The model's performance was validated by bootstrapping techniques.Results. A total of 316 patients were included. The median overall survival was 10 months. Multivariate Cox analysis yielded 5 prognostic variables of which the coefficients were included in the risk score. AgeConclusions. We developed a DIPG survival prediction tool that can be used to predict the outcome of patients and for stratification in trials. Validation of the model is needed in a prospective cohort.

KW - brainstem neoplasms

KW - glioma

KW - magnetic resonance imaging

KW - pons

KW - prognosis

KW - BRAIN-STEM GLIOMA

KW - CHILDHOOD

KW - TUMORS

KW - CHEMOTHERAPY

KW - TRIALS

U2 - 10.1093/neuonc/nou104

DO - 10.1093/neuonc/nou104

M3 - Article

SN - 1522-8517

VL - 17

SP - 160

EP - 166

JO - Neuro-Oncology

JF - Neuro-Oncology

IS - 1

ER -

Survival prediction model of children with diffuse intrinsic pontine glioma based on clinical and radiological criteria

Abstract

Keywords

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