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; Cacha 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; Dannis 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, Cacha 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, Dannis G van Vuurden

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

117 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 ≤3 years, longer symptom duration at diagnosis, and use of oral and intravenous chemotherapy were favorable predictors, while ring enhancement on MRI at diagnosis was an unfavorable predictor. With increasing risk score categories, overall survival decreased significantly. The model can distinguish between patients with very short, average, and increased overall survival (medians of 7.0, 9.7, and 13.7 mo, respectively). The area under the receiver operating characteristic curve was 0.68.

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)	160-6
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
Externally published	Yes

Keywords

Adolescent
Brain Stem Neoplasms/diagnosis
Child
Child, Preschool
Cohort Studies
Female
Glioma/diagnosis
Humans
Infant
Infant, Newborn
Magnetic Resonance Imaging
Male
Proportional Hazards Models
Retrospective Studies
Treatment Outcome

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10.1093/neuonc/nou104

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Jansen, M. H., Veldhuijzen van Zanten, S. E., Sanchez Aliaga, E., 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), 160-6. https://doi.org/10.1093/neuonc/nou104

@article{4984106d98df40288df96c6dcc2129e0,

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. Age ≤3 years, longer symptom duration at diagnosis, and use of oral and intravenous chemotherapy were favorable predictors, while ring enhancement on MRI at diagnosis was an unfavorable predictor. With increasing risk score categories, overall survival decreased significantly. The model can distinguish between patients with very short, average, and increased overall survival (medians of 7.0, 9.7, and 13.7 mo, respectively). The area under the receiver operating characteristic curve was 0.68.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.",

keywords = "Adolescent, Brain Stem Neoplasms/diagnosis, Child, Child, Preschool, Cohort Studies, Female, Glioma/diagnosis, Humans, Infant, Infant, Newborn, Magnetic Resonance Imaging, Male, Proportional Hazards Models, Retrospective Studies, Treatment Outcome",

author = "Jansen, {Marc H} and {Veldhuijzen van Zanten}, {Sophie E} and {Sanchez Aliaga}, Esther 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, {Cacha 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}, {Dannis G}",

year = "2015",

month = jan,

doi = "10.1093/neuonc/nou104",

language = "English",

volume = "17",

pages = "160--6",

journal = "Neuro-Oncology",

issn = "1522-8517",

publisher = "Oxford University Press",

number = "1",

}

Jansen, MH, Veldhuijzen van Zanten, SE, Sanchez Aliaga, E, 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. 160-6. 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 - Veldhuijzen van Zanten, Sophie E

AU - Sanchez Aliaga, Esther

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, Cacha 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, Dannis 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. Age ≤3 years, longer symptom duration at diagnosis, and use of oral and intravenous chemotherapy were favorable predictors, while ring enhancement on MRI at diagnosis was an unfavorable predictor. With increasing risk score categories, overall survival decreased significantly. The model can distinguish between patients with very short, average, and increased overall survival (medians of 7.0, 9.7, and 13.7 mo, respectively). The area under the receiver operating characteristic curve was 0.68.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.

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. Age ≤3 years, longer symptom duration at diagnosis, and use of oral and intravenous chemotherapy were favorable predictors, while ring enhancement on MRI at diagnosis was an unfavorable predictor. With increasing risk score categories, overall survival decreased significantly. The model can distinguish between patients with very short, average, and increased overall survival (medians of 7.0, 9.7, and 13.7 mo, respectively). The area under the receiver operating characteristic curve was 0.68.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.

KW - Adolescent

KW - Brain Stem Neoplasms/diagnosis

KW - Child

KW - Child, Preschool

KW - Cohort Studies

KW - Female

KW - Glioma/diagnosis

KW - Humans

KW - Infant

KW - Infant, Newborn

KW - Magnetic Resonance Imaging

KW - Male

KW - Proportional Hazards Models

KW - Retrospective Studies

KW - Treatment Outcome

UR - http://www.scopus.com/inward/record.url?scp=84922503066&partnerID=8YFLogxK

U2 - 10.1093/neuonc/nou104

DO - 10.1093/neuonc/nou104

M3 - Article

C2 - 24903904

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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