Local and systemic therapy of recurrent ependymoma in children and adolescents: short- and long-term results of the E-HIT-REZ 2005 study.
chemotherapy
ependymoma
radiotherapy
relapse
tumor resection
Journal
Neuro-oncology
ISSN: 1523-5866
Titre abrégé: Neuro Oncol
Pays: England
ID NLM: 100887420
Informations de publication
Date de publication:
01 06 2021
01 06 2021
Historique:
pubmed:
18
12
2020
medline:
22
6
2021
entrez:
17
12
2020
Statut:
ppublish
Résumé
Survival in recurrent ependymomas in children and adolescents mainly depends on the extent of resection. Studies on repeated radiotherapy and chemotherapy at relapse have shown conflicting results. Using data from the German multi-center E-HIT-REZ-2005 study, we examined the role of local therapy and the efficacy of chemotherapy with blockwise temozolomide (TMZ) in children and adolescents with recurrent ependymomas. Fifty-three patients with a median age of 6.9 years (1.25-25.4) at first recurrence and a median follow-up time of 36 months (2-115) were recruited. Gross- and near-total resection (GTR/NTR) were achieved in 34 (64.2%) patients and associated with a markedly improved 5-year overall survival (OS) of 48.7% vs. 5.3% in less than GTR/NTR. Radiotherapy showed no improvement in OS following complete resection (OS: 70 (CI: 19.9-120.1) vs. 95 (CI: 20.7-169.4) months), but an advantage was found in less than GTR/NTR (OS: 22 (CI: 12.7-31.3) vs. 7 (CI: 0-15.8) months). Following the application of TMZ, disease progression was observed in most evaluable cases (18/21). A subsequent change to oral etoposide and trofosfamide showed no improved response. PF-A EPN were most abundant in relapses (n = 27). RELA-positive EPN (n = 5) had a 5-year OS of 0%. The extent of resection is the most important predictor of survival at relapse. Focal re-irradiation is a useful approach if complete resection cannot be achieved, but no additional benefit was seen after GTR/NTR. Longer-term disease stabilization (>6 months) mediated by TMZ occurred in a small number of cases (14.3%).
Sections du résumé
BACKGROUND
Survival in recurrent ependymomas in children and adolescents mainly depends on the extent of resection. Studies on repeated radiotherapy and chemotherapy at relapse have shown conflicting results.
METHODS
Using data from the German multi-center E-HIT-REZ-2005 study, we examined the role of local therapy and the efficacy of chemotherapy with blockwise temozolomide (TMZ) in children and adolescents with recurrent ependymomas.
RESULTS
Fifty-three patients with a median age of 6.9 years (1.25-25.4) at first recurrence and a median follow-up time of 36 months (2-115) were recruited. Gross- and near-total resection (GTR/NTR) were achieved in 34 (64.2%) patients and associated with a markedly improved 5-year overall survival (OS) of 48.7% vs. 5.3% in less than GTR/NTR. Radiotherapy showed no improvement in OS following complete resection (OS: 70 (CI: 19.9-120.1) vs. 95 (CI: 20.7-169.4) months), but an advantage was found in less than GTR/NTR (OS: 22 (CI: 12.7-31.3) vs. 7 (CI: 0-15.8) months). Following the application of TMZ, disease progression was observed in most evaluable cases (18/21). A subsequent change to oral etoposide and trofosfamide showed no improved response. PF-A EPN were most abundant in relapses (n = 27). RELA-positive EPN (n = 5) had a 5-year OS of 0%.
CONCLUSION
The extent of resection is the most important predictor of survival at relapse. Focal re-irradiation is a useful approach if complete resection cannot be achieved, but no additional benefit was seen after GTR/NTR. Longer-term disease stabilization (>6 months) mediated by TMZ occurred in a small number of cases (14.3%).
Identifiants
pubmed: 33331885
pii: 6040794
doi: 10.1093/neuonc/noaa276
pmc: PMC8168820
doi:
Substances chimiques
Temozolomide
YF1K15M17Y
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
1012-1023Commentaires et corrections
Type : CommentIn
Informations de copyright
© The Author(s) 2020. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.
Références
Acta Neuropathol Commun. 2019 Nov 14;7(1):181
pubmed: 31727173
Childs Nerv Syst. 2005 Mar;21(3):221-6
pubmed: 15599561
Neuro Oncol. 2019 Mar 18;21(4):547-557
pubmed: 30452715
Cancer Cell. 2015 May 11;27(5):728-43
pubmed: 25965575
Brain Pathol. 2019 Jan;29(1):75-84
pubmed: 30417460
Clin Cancer Res. 2006 Apr 1;12(7 Pt 1):2070-9
pubmed: 16609018
Int J Radiat Oncol Biol Phys. 2012 Aug 1;83(5):1541-8
pubmed: 22245198
J Clin Oncol. 2016 Jul 20;34(21):2468-77
pubmed: 27269943
Int J Radiat Oncol Biol Phys. 1990 Dec;19(6):1497-502
pubmed: 2262372
Childs Nerv Syst. 2009 Oct;25(10):1283-91
pubmed: 19484246
J Negat Results Biomed. 2011 May 31;10:7
pubmed: 21627842
Acta Neuropathol. 2014 Apr;127(4):609-11
pubmed: 24562983
Acta Neuropathol. 2018 Aug;136(2):227-237
pubmed: 30019219
Neuro Oncol. 2016 Oct;18(10):1451-60
pubmed: 27194148
Pediatr Blood Cancer. 2014 Jul;61(7):1195-201
pubmed: 24615997
Int J Radiat Oncol Biol Phys. 2008 May 1;71(1):87-97
pubmed: 18406885
Mod Pathol. 2008 Feb;21(2):165-77
pubmed: 18084249
Neuro Oncol. 2019 Oct 9;21(10):1319-1330
pubmed: 30976811
Childs Nerv Syst. 2010 Jul;26(7):905-11
pubmed: 20039045
Acta Neuropathol. 2018 Aug;136(2):211-226
pubmed: 29909548
Eur J Cancer. 2009 Jul;45(10):1815-23
pubmed: 19427780
J Neurooncol. 2018 Nov;140(2):457-465
pubmed: 30109673
Int J Radiat Oncol Biol Phys. 2018 Feb 01;100(2):507-515
pubmed: 29229328
Acta Neuropathol. 2019 Dec;138(6):1075-1089
pubmed: 31414211
Childs Nerv Syst. 1999 Oct;15(10):563-70
pubmed: 10550587
Cancer Cell. 2011 Aug 16;20(2):143-57
pubmed: 21840481
Cancer. 2007 Oct 1;110(7):1542-50
pubmed: 17705175
Oncotarget. 2017 Dec 15;9(8):7822-7831
pubmed: 29487694
J Clin Oncol. 2019 Apr 20;37(12):974-983
pubmed: 30811284
Childs Nerv Syst. 2009 Oct;25(10):1293-301
pubmed: 19360417
J Neuropathol Exp Neurol. 2019 Sep 1;78(9):791-797
pubmed: 31373367
Am J Pathol. 2001 Mar;158(3):1137-43
pubmed: 11238062