Local and Systemic Therapy of Recurrent Ependymoma in Children and Adolescents: Short- and Long-term Results of the E-HIT-REZ 2005 Study
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Local and Systemic Therapy of Recurrent Ependymoma in Children and Adolescents: Short- and Long-term Results of the E-HIT-REZ 2005 Study. / Adolph, Jonas E; Fleischhack, Gudrun; Mikasch, Ruth; Zeller, Julia; Warmuth-Metz, Monika; Bison, Brigitte; Mynarek, Martin; Rutkowski, Stefan; Schüller, Ulrich; von Hoff, Katja; Obrecht, Denise; Pietsch, Torsten; Pfister, Stefan M; Pajtler, Kristian W; Witt, Olaf; Witt, Hendrik; Kortmann, Rolf-Dieter; Timmermann, Beate; Krauß, Jürgen; Frühwald, Michael C; Faldum, Andreas; Kwiecien, Robert; Bode, Udo; Tippelt, Stephan; German GPOH HIT-Network.
in: NEURO-ONCOLOGY, Jahrgang 23, Nr. 6, 01.06.2021, S. 1012-1023.Publikationen: SCORING: Beitrag in Fachzeitschrift/Zeitung › SCORING: Zeitschriftenaufsatz › Forschung › Begutachtung
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TY - JOUR
T1 - Local and Systemic Therapy of Recurrent Ependymoma in Children and Adolescents: Short- and Long-term Results of the E-HIT-REZ 2005 Study
AU - Adolph, Jonas E
AU - Fleischhack, Gudrun
AU - Mikasch, Ruth
AU - Zeller, Julia
AU - Warmuth-Metz, Monika
AU - Bison, Brigitte
AU - Mynarek, Martin
AU - Rutkowski, Stefan
AU - Schüller, Ulrich
AU - von Hoff, Katja
AU - Obrecht, Denise
AU - Pietsch, Torsten
AU - Pfister, Stefan M
AU - Pajtler, Kristian W
AU - Witt, Olaf
AU - Witt, Hendrik
AU - Kortmann, Rolf-Dieter
AU - Timmermann, Beate
AU - Krauß, Jürgen
AU - Frühwald, Michael C
AU - Faldum, Andreas
AU - Kwiecien, Robert
AU - Bode, Udo
AU - Tippelt, Stephan
AU - German GPOH HIT-Network
N1 - © 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.
PY - 2021/6/1
Y1 - 2021/6/1
N2 - 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%).
AB - 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%).
U2 - 10.1093/neuonc/noaa276
DO - 10.1093/neuonc/noaa276
M3 - SCORING: Journal article
C2 - 33331885
VL - 23
SP - 1012
EP - 1023
JO - NEURO-ONCOLOGY
JF - NEURO-ONCOLOGY
SN - 1522-8517
IS - 6
ER -