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MBCL-09. ISOLATED M1 METASTASES IN PEDIATRIC MEDULLOBLASTOMA: IS POSTOPERATIVE RADIOTHERAPY FOLLOWED BY MAINTENANCE CHEMOTHERAPY SUPERIOR TO POSTOPERATIVE SANDWICH-CHEMOTHERAPY AND RADIOTHERAPY?

BACKGROUND: Impact of isolated spread into the cerebrospinal fluid (CSF) is still not investigated comprehensively for childhood medulloblastoma and the best therapeutic strategy is currently unclear. MATERIAL AND METHODS: Sixty-six patients with isolated M1-MB registered to the HIT-MED-database fro...

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Autores principales: Obrecht, Denise, Mynarek, Martin, Hagel, Christian, Kwiecien, Robert, Spohn, Michael, Bockmayr, Michael L, Bison, Brigitte, Warmuth-Metz, Monika, Pfister, Stefan M, Jones, David T W, Sturm, Dominik, von Deimling, Andreas, Sahm, Felix, von Hoff, Katja, Juhnke, B - Ole, Benesch, Martin, Gerber, Nicolas U, von Bueren, André O, Friedrich, Carsten, Kortmann, Rolf-Dieter, Schwarz, Rudolf, Pietsch, Torsten, Rutkowski, Stefan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7715719/
http://dx.doi.org/10.1093/neuonc/noaa222.485
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author Obrecht, Denise
Mynarek, Martin
Hagel, Christian
Kwiecien, Robert
Spohn, Michael
Bockmayr, Michael L
Bison, Brigitte
Warmuth-Metz, Monika
Pfister, Stefan M
Jones, David T W
Sturm, Dominik
von Deimling, Andreas
Sahm, Felix
von Hoff, Katja
Juhnke, B - Ole
Benesch, Martin
Gerber, Nicolas U
von Bueren, André O
Friedrich, Carsten
Kortmann, Rolf-Dieter
Schwarz, Rudolf
Pietsch, Torsten
Rutkowski, Stefan
author_facet Obrecht, Denise
Mynarek, Martin
Hagel, Christian
Kwiecien, Robert
Spohn, Michael
Bockmayr, Michael L
Bison, Brigitte
Warmuth-Metz, Monika
Pfister, Stefan M
Jones, David T W
Sturm, Dominik
von Deimling, Andreas
Sahm, Felix
von Hoff, Katja
Juhnke, B - Ole
Benesch, Martin
Gerber, Nicolas U
von Bueren, André O
Friedrich, Carsten
Kortmann, Rolf-Dieter
Schwarz, Rudolf
Pietsch, Torsten
Rutkowski, Stefan
author_sort Obrecht, Denise
collection PubMed
description BACKGROUND: Impact of isolated spread into the cerebrospinal fluid (CSF) is still not investigated comprehensively for childhood medulloblastoma and the best therapeutic strategy is currently unclear. MATERIAL AND METHODS: Sixty-six patients with isolated M1-MB registered to the HIT-MED-database from 2000–2018 were identified. CSF and MRI were centrally reviewed for all patients. Patients were stratified by age and either treated with upfront craniospinal irradiation (CSI) followed by maintenance chemotherapy (CT) or with postoperative CT and delayed CSI. RESULTS: Forty-nine patients were non-infants ≥4 years and seventeen were infants <4 years. Median age was 7.3y (1.1–18.0). 83.3% were histologically classified as CMB, 12.1% as LCA-MB and 4.6% as DMB. Molecular subgroup was Gr.3 in 25.8%, Gr.4 in 28.8%, SHH in 4.5%, WNT in 1.5% and not evaluated for 39.4%. Lumbar puncture was performed on median postoperative day 19 (range: 14–77). Median follow-up for survivors was 7.6y (range: 1.2–15.9). The whole cohort showed a 3y- and 5y-PFS of 68.0(±6.0) and 60.0(±6.5)%, while OS was 79.1(±5.2) and 72.9(± 5.9)%. 10y-OS was 54.4(±7.5). Patients with upfront CSI had more favourable outcomes (5y-PFS 66.1 vs. 55.8% [p=0.119]; 5y-OS 90.6 vs. 64.5% [p=0.035]). The trend towards improved survival in patients with postoperative CSI was retained when only non-infants were considered (p(PFS)=0.176, p(OS)=0.055). M1-persistence occurred exclusively in patients with postoperative CT. CONCLUSION: Isolated M1-MB is rare. Patients without contraindication for CSI appear to benefit from treatment by upfront CSI followed by maintenance CT, while cumulative CT-doses would be reduced compared to sandwich strategies.
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spelling pubmed-77157192020-12-09 MBCL-09. ISOLATED M1 METASTASES IN PEDIATRIC MEDULLOBLASTOMA: IS POSTOPERATIVE RADIOTHERAPY FOLLOWED BY MAINTENANCE CHEMOTHERAPY SUPERIOR TO POSTOPERATIVE SANDWICH-CHEMOTHERAPY AND RADIOTHERAPY? Obrecht, Denise Mynarek, Martin Hagel, Christian Kwiecien, Robert Spohn, Michael Bockmayr, Michael L Bison, Brigitte Warmuth-Metz, Monika Pfister, Stefan M Jones, David T W Sturm, Dominik von Deimling, Andreas Sahm, Felix von Hoff, Katja Juhnke, B - Ole Benesch, Martin Gerber, Nicolas U von Bueren, André O Friedrich, Carsten Kortmann, Rolf-Dieter Schwarz, Rudolf Pietsch, Torsten Rutkowski, Stefan Neuro Oncol Medulloblastoma (Clinical) BACKGROUND: Impact of isolated spread into the cerebrospinal fluid (CSF) is still not investigated comprehensively for childhood medulloblastoma and the best therapeutic strategy is currently unclear. MATERIAL AND METHODS: Sixty-six patients with isolated M1-MB registered to the HIT-MED-database from 2000–2018 were identified. CSF and MRI were centrally reviewed for all patients. Patients were stratified by age and either treated with upfront craniospinal irradiation (CSI) followed by maintenance chemotherapy (CT) or with postoperative CT and delayed CSI. RESULTS: Forty-nine patients were non-infants ≥4 years and seventeen were infants <4 years. Median age was 7.3y (1.1–18.0). 83.3% were histologically classified as CMB, 12.1% as LCA-MB and 4.6% as DMB. Molecular subgroup was Gr.3 in 25.8%, Gr.4 in 28.8%, SHH in 4.5%, WNT in 1.5% and not evaluated for 39.4%. Lumbar puncture was performed on median postoperative day 19 (range: 14–77). Median follow-up for survivors was 7.6y (range: 1.2–15.9). The whole cohort showed a 3y- and 5y-PFS of 68.0(±6.0) and 60.0(±6.5)%, while OS was 79.1(±5.2) and 72.9(± 5.9)%. 10y-OS was 54.4(±7.5). Patients with upfront CSI had more favourable outcomes (5y-PFS 66.1 vs. 55.8% [p=0.119]; 5y-OS 90.6 vs. 64.5% [p=0.035]). The trend towards improved survival in patients with postoperative CSI was retained when only non-infants were considered (p(PFS)=0.176, p(OS)=0.055). M1-persistence occurred exclusively in patients with postoperative CT. CONCLUSION: Isolated M1-MB is rare. Patients without contraindication for CSI appear to benefit from treatment by upfront CSI followed by maintenance CT, while cumulative CT-doses would be reduced compared to sandwich strategies. Oxford University Press 2020-12-04 /pmc/articles/PMC7715719/ http://dx.doi.org/10.1093/neuonc/noaa222.485 Text en © The Author(s) 2020. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. http://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Medulloblastoma (Clinical)
Obrecht, Denise
Mynarek, Martin
Hagel, Christian
Kwiecien, Robert
Spohn, Michael
Bockmayr, Michael L
Bison, Brigitte
Warmuth-Metz, Monika
Pfister, Stefan M
Jones, David T W
Sturm, Dominik
von Deimling, Andreas
Sahm, Felix
von Hoff, Katja
Juhnke, B - Ole
Benesch, Martin
Gerber, Nicolas U
von Bueren, André O
Friedrich, Carsten
Kortmann, Rolf-Dieter
Schwarz, Rudolf
Pietsch, Torsten
Rutkowski, Stefan
MBCL-09. ISOLATED M1 METASTASES IN PEDIATRIC MEDULLOBLASTOMA: IS POSTOPERATIVE RADIOTHERAPY FOLLOWED BY MAINTENANCE CHEMOTHERAPY SUPERIOR TO POSTOPERATIVE SANDWICH-CHEMOTHERAPY AND RADIOTHERAPY?
title MBCL-09. ISOLATED M1 METASTASES IN PEDIATRIC MEDULLOBLASTOMA: IS POSTOPERATIVE RADIOTHERAPY FOLLOWED BY MAINTENANCE CHEMOTHERAPY SUPERIOR TO POSTOPERATIVE SANDWICH-CHEMOTHERAPY AND RADIOTHERAPY?
title_full MBCL-09. ISOLATED M1 METASTASES IN PEDIATRIC MEDULLOBLASTOMA: IS POSTOPERATIVE RADIOTHERAPY FOLLOWED BY MAINTENANCE CHEMOTHERAPY SUPERIOR TO POSTOPERATIVE SANDWICH-CHEMOTHERAPY AND RADIOTHERAPY?
title_fullStr MBCL-09. ISOLATED M1 METASTASES IN PEDIATRIC MEDULLOBLASTOMA: IS POSTOPERATIVE RADIOTHERAPY FOLLOWED BY MAINTENANCE CHEMOTHERAPY SUPERIOR TO POSTOPERATIVE SANDWICH-CHEMOTHERAPY AND RADIOTHERAPY?
title_full_unstemmed MBCL-09. ISOLATED M1 METASTASES IN PEDIATRIC MEDULLOBLASTOMA: IS POSTOPERATIVE RADIOTHERAPY FOLLOWED BY MAINTENANCE CHEMOTHERAPY SUPERIOR TO POSTOPERATIVE SANDWICH-CHEMOTHERAPY AND RADIOTHERAPY?
title_short MBCL-09. ISOLATED M1 METASTASES IN PEDIATRIC MEDULLOBLASTOMA: IS POSTOPERATIVE RADIOTHERAPY FOLLOWED BY MAINTENANCE CHEMOTHERAPY SUPERIOR TO POSTOPERATIVE SANDWICH-CHEMOTHERAPY AND RADIOTHERAPY?
title_sort mbcl-09. isolated m1 metastases in pediatric medulloblastoma: is postoperative radiotherapy followed by maintenance chemotherapy superior to postoperative sandwich-chemotherapy and radiotherapy?
topic Medulloblastoma (Clinical)
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7715719/
http://dx.doi.org/10.1093/neuonc/noaa222.485
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