TY - JOUR
T1 - Survival benefit for patients with diffuse intrinsic pontine glioma (DIPG) undergoing re-irradiation at first progression
T2 - 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
N1 - Copyright © 2017 Elsevier Ltd. All rights reserved.
PY - 2017/3
Y1 - 2017/3
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 ≥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 < .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 < .01) and re-irradiation (corrected hazard ratio = .18-.22; P < .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 ≥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 < .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 < .01) and re-irradiation (corrected hazard ratio = .18-.22; P < .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 - Adolescent
KW - Brain Stem Neoplasms/mortality
KW - Child
KW - Child, Preschool
KW - Disease Progression
KW - Female
KW - Glioma/mortality
KW - Humans
KW - Magnetic Resonance Imaging
KW - Male
KW - Multivariate Analysis
KW - Prognosis
KW - Re-Irradiation/statistics & numerical data
KW - Retrospective Studies
KW - Survival Analysis
UR - http://www.scopus.com/inward/record.url?scp=85012281778&partnerID=8YFLogxK
U2 - 10.1016/j.ejca.2016.12.007
DO - 10.1016/j.ejca.2016.12.007
M3 - Article
C2 - 28161497
SN - 1879-0852
VL - 73
SP - 38
EP - 47
JO - European Journal of Cancer
JF - European Journal of Cancer
ER -