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Systemic chemotherapy of pediatric recurrent ependymomas: results from the German HIT-REZ studies
PURPOSE: Survival in recurrent ependymoma (EPN) depends mainly on the extent of resection achieved. When complete resection is not feasible, chemotherapy is often used to extend progression-free and overall survival. However, no consistent effect of chemotherapy on survival has been found in patient...
Autores principales: | , , , , , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Springer US
2021
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8585796/ https://www.ncbi.nlm.nih.gov/pubmed/34657224 http://dx.doi.org/10.1007/s11060-021-03867-8 |
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author | Adolph, Jonas E. Fleischhack, Gudrun Gaab, Christine Mikasch, Ruth Mynarek, Martin Rutkowski, Stefan Schüller, Ulrich Pfister, Stefan M. Pajtler, Kristian W. Milde, Till Witt, Olaf Bison, Brigitte Warmuth-Metz, Monika Kortmann, Rolf-Dieter Dietzsch, Stefan Pietsch, Torsten Timmermann, Beate Tippelt, Stephan |
author_facet | Adolph, Jonas E. Fleischhack, Gudrun Gaab, Christine Mikasch, Ruth Mynarek, Martin Rutkowski, Stefan Schüller, Ulrich Pfister, Stefan M. Pajtler, Kristian W. Milde, Till Witt, Olaf Bison, Brigitte Warmuth-Metz, Monika Kortmann, Rolf-Dieter Dietzsch, Stefan Pietsch, Torsten Timmermann, Beate Tippelt, Stephan |
author_sort | Adolph, Jonas E. |
collection | PubMed |
description | PURPOSE: Survival in recurrent ependymoma (EPN) depends mainly on the extent of resection achieved. When complete resection is not feasible, chemotherapy is often used to extend progression-free and overall survival. However, no consistent effect of chemotherapy on survival has been found in patients with recurrent EPN. METHODS: Systemic chemotherapeutic treatment of 138 patients enrolled in the German HIT-REZ-studies was analyzed. Survival depending on the use of chemotherapy, disease-stabilization rates (RR), duration of response (DOR) and time to progression (TTP) were estimated. RESULTS: Median age at first recurrence was 7.6 years (IQR: 4.0–13.6). At first recurrence, median PFS and OS were 15.3 (CI 13.3–20.0) and 36.9 months (CI 29.7–53.4), respectively. The Hazard Ratio for the use of chemotherapy in local recurrences in a time-dependent Cox-regression analysis was 0.99 (CI 0.74–1.33). Evaluable responses for 140 applied chemotherapies were analyzed, of which sirolimus showed the best RR (50%) and longest median TTP [11.51 (CI 3.98; 14.0) months] in nine patients, with the strongest impact found when sirolimus was used as a monotherapy. Seven patients with progression-free survival > 12 months after subtotal/no-resection facilitated by chemotherapy were found. No definitive survival advantage for any drug in a specific molecularly defined EPN type was found. CONCLUSION: No survival advantage for the general use of chemotherapy in recurrent EPN was found. In cases with incomplete resection, chemotherapy was able to extend survival in individual cases. Sirolimus showed the best RR, DOR and TTP out of all drugs analyzed and may warrant further investigation. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s11060-021-03867-8. |
format | Online Article Text |
id | pubmed-8585796 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Springer US |
record_format | MEDLINE/PubMed |
spelling | pubmed-85857962021-11-15 Systemic chemotherapy of pediatric recurrent ependymomas: results from the German HIT-REZ studies Adolph, Jonas E. Fleischhack, Gudrun Gaab, Christine Mikasch, Ruth Mynarek, Martin Rutkowski, Stefan Schüller, Ulrich Pfister, Stefan M. Pajtler, Kristian W. Milde, Till Witt, Olaf Bison, Brigitte Warmuth-Metz, Monika Kortmann, Rolf-Dieter Dietzsch, Stefan Pietsch, Torsten Timmermann, Beate Tippelt, Stephan J Neurooncol Clinical Study PURPOSE: Survival in recurrent ependymoma (EPN) depends mainly on the extent of resection achieved. When complete resection is not feasible, chemotherapy is often used to extend progression-free and overall survival. However, no consistent effect of chemotherapy on survival has been found in patients with recurrent EPN. METHODS: Systemic chemotherapeutic treatment of 138 patients enrolled in the German HIT-REZ-studies was analyzed. Survival depending on the use of chemotherapy, disease-stabilization rates (RR), duration of response (DOR) and time to progression (TTP) were estimated. RESULTS: Median age at first recurrence was 7.6 years (IQR: 4.0–13.6). At first recurrence, median PFS and OS were 15.3 (CI 13.3–20.0) and 36.9 months (CI 29.7–53.4), respectively. The Hazard Ratio for the use of chemotherapy in local recurrences in a time-dependent Cox-regression analysis was 0.99 (CI 0.74–1.33). Evaluable responses for 140 applied chemotherapies were analyzed, of which sirolimus showed the best RR (50%) and longest median TTP [11.51 (CI 3.98; 14.0) months] in nine patients, with the strongest impact found when sirolimus was used as a monotherapy. Seven patients with progression-free survival > 12 months after subtotal/no-resection facilitated by chemotherapy were found. No definitive survival advantage for any drug in a specific molecularly defined EPN type was found. CONCLUSION: No survival advantage for the general use of chemotherapy in recurrent EPN was found. In cases with incomplete resection, chemotherapy was able to extend survival in individual cases. Sirolimus showed the best RR, DOR and TTP out of all drugs analyzed and may warrant further investigation. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s11060-021-03867-8. Springer US 2021-10-16 2021 /pmc/articles/PMC8585796/ /pubmed/34657224 http://dx.doi.org/10.1007/s11060-021-03867-8 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . |
spellingShingle | Clinical Study Adolph, Jonas E. Fleischhack, Gudrun Gaab, Christine Mikasch, Ruth Mynarek, Martin Rutkowski, Stefan Schüller, Ulrich Pfister, Stefan M. Pajtler, Kristian W. Milde, Till Witt, Olaf Bison, Brigitte Warmuth-Metz, Monika Kortmann, Rolf-Dieter Dietzsch, Stefan Pietsch, Torsten Timmermann, Beate Tippelt, Stephan Systemic chemotherapy of pediatric recurrent ependymomas: results from the German HIT-REZ studies |
title | Systemic chemotherapy of pediatric recurrent ependymomas: results from the German HIT-REZ studies |
title_full | Systemic chemotherapy of pediatric recurrent ependymomas: results from the German HIT-REZ studies |
title_fullStr | Systemic chemotherapy of pediatric recurrent ependymomas: results from the German HIT-REZ studies |
title_full_unstemmed | Systemic chemotherapy of pediatric recurrent ependymomas: results from the German HIT-REZ studies |
title_short | Systemic chemotherapy of pediatric recurrent ependymomas: results from the German HIT-REZ studies |
title_sort | systemic chemotherapy of pediatric recurrent ependymomas: results from the german hit-rez studies |
topic | Clinical Study |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8585796/ https://www.ncbi.nlm.nih.gov/pubmed/34657224 http://dx.doi.org/10.1007/s11060-021-03867-8 |
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