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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

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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, Vol. 23, No. 6, 01.06.2021, p. 1012-1023.

Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review

Harvard

Adolph, JE, Fleischhack, G, Mikasch, R, Zeller, J, Warmuth-Metz, M, Bison, B, Mynarek, M, Rutkowski, S, Schüller, U, von Hoff, K, Obrecht, D, Pietsch, T, Pfister, SM, Pajtler, KW, Witt, O, Witt, H, Kortmann, R-D, Timmermann, B, Krauß, J, Frühwald, MC, Faldum, A, Kwiecien, R, Bode, U, Tippelt, S & German GPOH HIT-Network 2021, 'Local and Systemic Therapy of Recurrent Ependymoma in Children and Adolescents: Short- and Long-term Results of the E-HIT-REZ 2005 Study', NEURO-ONCOLOGY, vol. 23, no. 6, pp. 1012-1023. https://doi.org/10.1093/neuonc/noaa276

APA

Adolph, J. E., Fleischhack, G., Mikasch, R., Zeller, J., Warmuth-Metz, M., Bison, B., Mynarek, M., Rutkowski, S., Schüller, U., von Hoff, K., Obrecht, D., Pietsch, T., Pfister, S. M., Pajtler, K. W., Witt, O., Witt, H., Kortmann, R-D., Timmermann, B., Krauß, J., ... German GPOH HIT-Network (2021). Local and Systemic Therapy of Recurrent Ependymoma in Children and Adolescents: Short- and Long-term Results of the E-HIT-REZ 2005 Study. NEURO-ONCOLOGY, 23(6), 1012-1023. https://doi.org/10.1093/neuonc/noaa276

Vancouver

Adolph JE, Fleischhack G, Mikasch R, Zeller J, Warmuth-Metz M, Bison B et al. Local and Systemic Therapy of Recurrent Ependymoma in Children and Adolescents: Short- and Long-term Results of the E-HIT-REZ 2005 Study. NEURO-ONCOLOGY. 2021 Jun 1;23(6):1012-1023. https://doi.org/10.1093/neuonc/noaa276

Bibtex

@article{fc35034ac5c64bdd9c53f4ee775e0752,
title = "Local and Systemic Therapy of Recurrent Ependymoma in Children and Adolescents: Short- and Long-term Results of the E-HIT-REZ 2005 Study",
abstract = "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%).",
author = "Adolph, {Jonas E} and Gudrun Fleischhack and Ruth Mikasch and Julia Zeller and Monika Warmuth-Metz and Brigitte Bison and Martin Mynarek and Stefan Rutkowski and Ulrich Sch{\"u}ller and {von Hoff}, Katja and Denise Obrecht and Torsten Pietsch and Pfister, {Stefan M} and Pajtler, {Kristian W} and Olaf Witt and Hendrik Witt and Rolf-Dieter Kortmann and Beate Timmermann and J{\"u}rgen Krau{\ss} and Fr{\"u}hwald, {Michael C} and Andreas Faldum and Robert Kwiecien and Udo Bode and Stephan Tippelt and {German GPOH HIT-Network}",
note = "{\textcopyright} 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.",
year = "2021",
month = jun,
day = "1",
doi = "10.1093/neuonc/noaa276",
language = "English",
volume = "23",
pages = "1012--1023",
journal = "NEURO-ONCOLOGY",
issn = "1522-8517",
publisher = "Oxford University Press",
number = "6",

}

RIS

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 -