Survival benefit for patients with diffuse intrinsic pontine glioma (DIPG) undergoing re-irradiation at first progression: A matched-cohort analysis on behalf of the SIOP-E-HGG/DIPG working group.

Geert O. Janssens, Lorenza Gandola, Stephanie Bolle, Henry Mandeville, Monica Ramos-Albiac, Karen van Beek, Helen Benghiat, Bianca Hoeben, Andres Morales La Madrid, Rolf Dieter Kortmann, Darren Hargrave, Johan Menten, Emilia Pecori, Veronica Biassoni, Andre O. von Bueren, Dannis G van Vuurden, Maura Massimino, Dominik Sturm, Max Peters, Christof M. Kramm

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Abstract

BACKGROUND: Overall survival (OS) of patients with diffuse intrinsic pontine glioma (DIPG) is poor. The purpose of this study is to analyse benefit and toxicity of re-irradiation at first progression. METHODS: At first progression, 31 children with DIPG, aged 2-16 years, underwent re-irradiation (dose 19.8-30.0 Gy) alone (n = 16) or combined with systemic therapy (n = 15). At initial presentation, all patients had typical symptoms and characteristic MRI features of DIPG, or biopsy-proven high-grade glioma. An interval of textgreater/=3 months after upfront radiotherapy was required before re-irradiation. Thirty-nine patients fulfilling the same criteria receiving radiotherapy at diagnosis, followed by best supportive care (n = 20) or systemic therapy (n = 19) at progression but no re-irradiation, were eligible for a matched-cohort analysis. RESULTS: Median OS for patients undergoing re-irradiation was 13.7 months. For a similar median progression-free survival after upfront radiotherapy (8.2 versus 7.7 months; P = .58), a significant benefit in median OS (13.7 versus 10.3 months; P = .04) was observed in favour of patients undergoing re-irradiation. Survival benefit of re-irradiation increased with a longer interval between end-of-radiotherapy and first progression (3-6 months: 4.0 versus 2.7; P textless .01; 6-12 months: 6.4 versus 3.3; P = .04). Clinical improvement with re-irradiation was observed in 24/31 (77 patients. No grade 4-5 toxicity was recorded. On multivariable analysis, interval to progression (corrected hazard ratio = .27-.54; P textless .01) and re-irradiation (corrected hazard ratio = .18-.22; P textless .01) remained prognostic for survival. A risk score (RS), comprising 5 categories, was developed to predict survival from first progression (ROC: .79). Median survival ranges from 1.0 month (RS-1) to 6.7 months (RS-5). CONCLUSIONS: The majority of patients with DIPG, responding to upfront radiotherapy, do benefit of re-irradiation with acceptable tolerability.
Original languageUndefined/Unknown
Pages (from-to)38-47
Number of pages10
JournalEuropean Journal of Cancer
Volume73
DOIs
Publication statusPublished - Mar 1 2017

Keywords

  • Diffuse intrinsic pontine glioma (DIPG), Matched-cohort analysis, radiotherapy, Re-irradiation, Survival prediction model

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Survival benefit for patients with diffuse intrinsic pontine glioma (DIPG) undergoing re-irradiation at first progression: A matched-cohort analysis on behalf of the SIOP-E-HGG/DIPG working group. / Janssens, Geert O.; Gandola, Lorenza; Bolle, Stephanie; Mandeville, Henry; Ramos-Albiac, Monica; van Beek, Karen; Benghiat, Helen; Hoeben, Bianca; Morales La Madrid, Andres; Kortmann, Rolf Dieter; Hargrave, Darren; Menten, Johan; Pecori, Emilia; Biassoni, Veronica; von Bueren, Andre O.; van Vuurden, Dannis G; Massimino, Maura; Sturm, Dominik; Peters, Max; Kramm, Christof M.

In: European Journal of Cancer, Vol. 73, 01.03.2017, p. 38-47.

Research output: Contribution to journalArticle

Janssens, GO, Gandola, L, Bolle, S, Mandeville, H, Ramos-Albiac, M, van Beek, K, Benghiat, H, Hoeben, B, Morales La Madrid, A, Kortmann, RD, Hargrave, D, Menten, J, Pecori, E, Biassoni, V, von Bueren, AO, van Vuurden, DG, Massimino, M, Sturm, D, Peters, M & Kramm, CM 2017, 'Survival benefit for patients with diffuse intrinsic pontine glioma (DIPG) undergoing re-irradiation at first progression: A matched-cohort analysis on behalf of the SIOP-E-HGG/DIPG working group.', European Journal of Cancer, vol. 73, pp. 38-47. https://doi.org/10.1016/j.ejca.2016.12.007
Janssens, Geert O. ; Gandola, Lorenza ; Bolle, Stephanie ; Mandeville, Henry ; Ramos-Albiac, Monica ; van Beek, Karen ; Benghiat, Helen ; Hoeben, Bianca ; Morales La Madrid, Andres ; Kortmann, Rolf Dieter ; Hargrave, Darren ; Menten, Johan ; Pecori, Emilia ; Biassoni, Veronica ; von Bueren, Andre O. ; van Vuurden, Dannis G ; Massimino, Maura ; Sturm, Dominik ; Peters, Max ; Kramm, Christof M. / Survival benefit for patients with diffuse intrinsic pontine glioma (DIPG) undergoing re-irradiation at first progression: A matched-cohort analysis on behalf of the SIOP-E-HGG/DIPG working group. In: European Journal of Cancer. 2017 ; Vol. 73. pp. 38-47.
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title = "Survival benefit for patients with diffuse intrinsic pontine glioma (DIPG) undergoing re-irradiation at first progression: A matched-cohort analysis on behalf of the SIOP-E-HGG/DIPG working group.",
abstract = "BACKGROUND: Overall survival (OS) of patients with diffuse intrinsic pontine glioma (DIPG) is poor. The purpose of this study is to analyse benefit and toxicity of re-irradiation at first progression. METHODS: At first progression, 31 children with DIPG, aged 2-16 years, underwent re-irradiation (dose 19.8-30.0 Gy) alone (n = 16) or combined with systemic therapy (n = 15). At initial presentation, all patients had typical symptoms and characteristic MRI features of DIPG, or biopsy-proven high-grade glioma. An interval of textgreater/=3 months after upfront radiotherapy was required before re-irradiation. Thirty-nine patients fulfilling the same criteria receiving radiotherapy at diagnosis, followed by best supportive care (n = 20) or systemic therapy (n = 19) at progression but no re-irradiation, were eligible for a matched-cohort analysis. RESULTS: Median OS for patients undergoing re-irradiation was 13.7 months. For a similar median progression-free survival after upfront radiotherapy (8.2 versus 7.7 months; P = .58), a significant benefit in median OS (13.7 versus 10.3 months; P = .04) was observed in favour of patients undergoing re-irradiation. Survival benefit of re-irradiation increased with a longer interval between end-of-radiotherapy and first progression (3-6 months: 4.0 versus 2.7; P textless .01; 6-12 months: 6.4 versus 3.3; P = .04). Clinical improvement with re-irradiation was observed in 24/31 (77 patients. No grade 4-5 toxicity was recorded. On multivariable analysis, interval to progression (corrected hazard ratio = .27-.54; P textless .01) and re-irradiation (corrected hazard ratio = .18-.22; P textless .01) remained prognostic for survival. A risk score (RS), comprising 5 categories, was developed to predict survival from first progression (ROC: .79). Median survival ranges from 1.0 month (RS-1) to 6.7 months (RS-5). CONCLUSIONS: The majority of patients with DIPG, responding to upfront radiotherapy, do benefit of re-irradiation with acceptable tolerability.",
keywords = "Diffuse intrinsic pontine glioma (DIPG), Matched-cohort analysis, radiotherapy, Re-irradiation, Survival prediction model",
author = "Janssens, {Geert O.} and Lorenza Gandola and Stephanie Bolle and Henry Mandeville and Monica Ramos-Albiac and {van Beek}, Karen and Helen Benghiat and Bianca Hoeben and {Morales La Madrid}, Andres and Kortmann, {Rolf Dieter} and Darren Hargrave and Johan Menten and Emilia Pecori and Veronica Biassoni and {von Bueren}, {Andre O.} and {van Vuurden}, {Dannis G} and Maura Massimino and Dominik Sturm and Max Peters and Kramm, {Christof M.}",
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doi = "10.1016/j.ejca.2016.12.007",
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volume = "73",
pages = "38--47",
journal = "European Journal of Cancer",
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TY - JOUR

T1 - Survival benefit for patients with diffuse intrinsic pontine glioma (DIPG) undergoing re-irradiation at first progression: A matched-cohort analysis on behalf of the SIOP-E-HGG/DIPG working group.

AU - Janssens, Geert O.

AU - Gandola, Lorenza

AU - Bolle, Stephanie

AU - Mandeville, Henry

AU - Ramos-Albiac, Monica

AU - van Beek, Karen

AU - Benghiat, Helen

AU - Hoeben, Bianca

AU - Morales La Madrid, Andres

AU - Kortmann, Rolf Dieter

AU - Hargrave, Darren

AU - Menten, Johan

AU - Pecori, Emilia

AU - Biassoni, Veronica

AU - von Bueren, Andre O.

AU - van Vuurden, Dannis G

AU - Massimino, Maura

AU - Sturm, Dominik

AU - Peters, Max

AU - Kramm, Christof M.

PY - 2017/3/1

Y1 - 2017/3/1

N2 - BACKGROUND: Overall survival (OS) of patients with diffuse intrinsic pontine glioma (DIPG) is poor. The purpose of this study is to analyse benefit and toxicity of re-irradiation at first progression. METHODS: At first progression, 31 children with DIPG, aged 2-16 years, underwent re-irradiation (dose 19.8-30.0 Gy) alone (n = 16) or combined with systemic therapy (n = 15). At initial presentation, all patients had typical symptoms and characteristic MRI features of DIPG, or biopsy-proven high-grade glioma. An interval of textgreater/=3 months after upfront radiotherapy was required before re-irradiation. Thirty-nine patients fulfilling the same criteria receiving radiotherapy at diagnosis, followed by best supportive care (n = 20) or systemic therapy (n = 19) at progression but no re-irradiation, were eligible for a matched-cohort analysis. RESULTS: Median OS for patients undergoing re-irradiation was 13.7 months. For a similar median progression-free survival after upfront radiotherapy (8.2 versus 7.7 months; P = .58), a significant benefit in median OS (13.7 versus 10.3 months; P = .04) was observed in favour of patients undergoing re-irradiation. Survival benefit of re-irradiation increased with a longer interval between end-of-radiotherapy and first progression (3-6 months: 4.0 versus 2.7; P textless .01; 6-12 months: 6.4 versus 3.3; P = .04). Clinical improvement with re-irradiation was observed in 24/31 (77 patients. No grade 4-5 toxicity was recorded. On multivariable analysis, interval to progression (corrected hazard ratio = .27-.54; P textless .01) and re-irradiation (corrected hazard ratio = .18-.22; P textless .01) remained prognostic for survival. A risk score (RS), comprising 5 categories, was developed to predict survival from first progression (ROC: .79). Median survival ranges from 1.0 month (RS-1) to 6.7 months (RS-5). CONCLUSIONS: The majority of patients with DIPG, responding to upfront radiotherapy, do benefit of re-irradiation with acceptable tolerability.

AB - BACKGROUND: Overall survival (OS) of patients with diffuse intrinsic pontine glioma (DIPG) is poor. The purpose of this study is to analyse benefit and toxicity of re-irradiation at first progression. METHODS: At first progression, 31 children with DIPG, aged 2-16 years, underwent re-irradiation (dose 19.8-30.0 Gy) alone (n = 16) or combined with systemic therapy (n = 15). At initial presentation, all patients had typical symptoms and characteristic MRI features of DIPG, or biopsy-proven high-grade glioma. An interval of textgreater/=3 months after upfront radiotherapy was required before re-irradiation. Thirty-nine patients fulfilling the same criteria receiving radiotherapy at diagnosis, followed by best supportive care (n = 20) or systemic therapy (n = 19) at progression but no re-irradiation, were eligible for a matched-cohort analysis. RESULTS: Median OS for patients undergoing re-irradiation was 13.7 months. For a similar median progression-free survival after upfront radiotherapy (8.2 versus 7.7 months; P = .58), a significant benefit in median OS (13.7 versus 10.3 months; P = .04) was observed in favour of patients undergoing re-irradiation. Survival benefit of re-irradiation increased with a longer interval between end-of-radiotherapy and first progression (3-6 months: 4.0 versus 2.7; P textless .01; 6-12 months: 6.4 versus 3.3; P = .04). Clinical improvement with re-irradiation was observed in 24/31 (77 patients. No grade 4-5 toxicity was recorded. On multivariable analysis, interval to progression (corrected hazard ratio = .27-.54; P textless .01) and re-irradiation (corrected hazard ratio = .18-.22; P textless .01) remained prognostic for survival. A risk score (RS), comprising 5 categories, was developed to predict survival from first progression (ROC: .79). Median survival ranges from 1.0 month (RS-1) to 6.7 months (RS-5). CONCLUSIONS: The majority of patients with DIPG, responding to upfront radiotherapy, do benefit of re-irradiation with acceptable tolerability.

KW - Diffuse intrinsic pontine glioma (DIPG), Matched-cohort analysis, radiotherapy, Re-irradiation, Survival prediction model

U2 - 10.1016/j.ejca.2016.12.007

DO - 10.1016/j.ejca.2016.12.007

M3 - Articolo

VL - 73

SP - 38

EP - 47

JO - European Journal of Cancer

JF - European Journal of Cancer

SN - 0959-8049

ER -