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MBCL-24. CAN YOUNG CHILDREN WITH RELAPSED MEDULLOBLASTOMA BE SALVAGED AFTER INITIAL IRRADIATION-SPARING APPROACHES?

INTRODUCTION: Irradiation-sparing approaches are used in young children with medulloblastoma (MB) given the vulnerability of the developing brain to neurocognitive impairment. Limited data are available following relapse for these patients. We aimed to describe the management and outcomes of young c...

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Autores principales: Erker, Craig, Larouche, Valérie, Margol, Ashley, Cacciotti, Chantel, Perreault, Sébastien, Cohen, Kenneth J, AbdelBaki, Mohamed S, Hukin, Juliette, Rassekh, Shahrad Rod, Eisenstat, David D, Wilson, Beverly, Knipstein, Jeffrey, Hoppmann, Anna L, Sandler, Eric S, Dorris, Kathleen, Fay-McClymont, Taryn B, Salloum, Ralph, Harrod, Virginia L, Crooks, Bruce, Finlay, Jonathan L, Bouffet, Eric, Lafay-Cousin, Lucie
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7715107/
http://dx.doi.org/10.1093/neuonc/noaa222.500
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author Erker, Craig
Larouche, Valérie
Margol, Ashley
Cacciotti, Chantel
Perreault, Sébastien
Cohen, Kenneth J
AbdelBaki, Mohamed S
Hukin, Juliette
Rassekh, Shahrad Rod
Eisenstat, David D
Wilson, Beverly
Knipstein, Jeffrey
Hoppmann, Anna L
Sandler, Eric S
Dorris, Kathleen
Fay-McClymont, Taryn B
Salloum, Ralph
Harrod, Virginia L
Crooks, Bruce
Finlay, Jonathan L
Bouffet, Eric
Lafay-Cousin, Lucie
author_facet Erker, Craig
Larouche, Valérie
Margol, Ashley
Cacciotti, Chantel
Perreault, Sébastien
Cohen, Kenneth J
AbdelBaki, Mohamed S
Hukin, Juliette
Rassekh, Shahrad Rod
Eisenstat, David D
Wilson, Beverly
Knipstein, Jeffrey
Hoppmann, Anna L
Sandler, Eric S
Dorris, Kathleen
Fay-McClymont, Taryn B
Salloum, Ralph
Harrod, Virginia L
Crooks, Bruce
Finlay, Jonathan L
Bouffet, Eric
Lafay-Cousin, Lucie
author_sort Erker, Craig
collection PubMed
description INTRODUCTION: Irradiation-sparing approaches are used in young children with medulloblastoma (MB) given the vulnerability of the developing brain to neurocognitive impairment. Limited data are available following relapse for these patients. We aimed to describe the management and outcomes of young children with MB who relapsed after initial treatment without craniospinal irradiation (CSI). METHODS: International retrospective study including patients with MB diagnosed between 1995–2017, ≤ 72 months old, initially treated without CSI, who subsequently relapsed. RESULTS: Data are available for 52 patients (32 male). Median age at initial diagnosis was 27 months (range, 6–72) with 24 being metastatic. Initial therapy included conventional chemotherapy alone or high-dose chemotherapy (HDC) in 21 and 31 subjects, respectively. Three received upfront focal irradiation. Molecular subgrouping, available for 24 tumors, included 9 SHH and 15 non-WNT/non-SHH. Median time to relapse was 13 months (range, 3–63). Relapse was local, disseminated or combined in 20, 15, and 16, respectively. Salvage therapy with curative intent was given in 42/52 patients, including CSI in 28 subjects (median dose 36Gy, 18–41.4) or focal irradiation in 5 others. Three received HDC only. At a median follow-up time of 46 months (range, 4–255), 25 (48%) were alive, including 7/9 SHH and 7/15 non-WNT/non-SHH. The 2- and 5-year OS was 67% and 56% (SE, 7%), respectively. Two of 3 patients with SHH who did not receive salvage radiotherapy are survivors. CONCLUSION: A substantial proportion of young children who relapse following irradiation-sparing strategies can be salvaged. Neurocognitive and ototoxicity outcomes are being evaluated.
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spelling pubmed-77151072020-12-09 MBCL-24. CAN YOUNG CHILDREN WITH RELAPSED MEDULLOBLASTOMA BE SALVAGED AFTER INITIAL IRRADIATION-SPARING APPROACHES? Erker, Craig Larouche, Valérie Margol, Ashley Cacciotti, Chantel Perreault, Sébastien Cohen, Kenneth J AbdelBaki, Mohamed S Hukin, Juliette Rassekh, Shahrad Rod Eisenstat, David D Wilson, Beverly Knipstein, Jeffrey Hoppmann, Anna L Sandler, Eric S Dorris, Kathleen Fay-McClymont, Taryn B Salloum, Ralph Harrod, Virginia L Crooks, Bruce Finlay, Jonathan L Bouffet, Eric Lafay-Cousin, Lucie Neuro Oncol Medulloblastoma (Clinical) INTRODUCTION: Irradiation-sparing approaches are used in young children with medulloblastoma (MB) given the vulnerability of the developing brain to neurocognitive impairment. Limited data are available following relapse for these patients. We aimed to describe the management and outcomes of young children with MB who relapsed after initial treatment without craniospinal irradiation (CSI). METHODS: International retrospective study including patients with MB diagnosed between 1995–2017, ≤ 72 months old, initially treated without CSI, who subsequently relapsed. RESULTS: Data are available for 52 patients (32 male). Median age at initial diagnosis was 27 months (range, 6–72) with 24 being metastatic. Initial therapy included conventional chemotherapy alone or high-dose chemotherapy (HDC) in 21 and 31 subjects, respectively. Three received upfront focal irradiation. Molecular subgrouping, available for 24 tumors, included 9 SHH and 15 non-WNT/non-SHH. Median time to relapse was 13 months (range, 3–63). Relapse was local, disseminated or combined in 20, 15, and 16, respectively. Salvage therapy with curative intent was given in 42/52 patients, including CSI in 28 subjects (median dose 36Gy, 18–41.4) or focal irradiation in 5 others. Three received HDC only. At a median follow-up time of 46 months (range, 4–255), 25 (48%) were alive, including 7/9 SHH and 7/15 non-WNT/non-SHH. The 2- and 5-year OS was 67% and 56% (SE, 7%), respectively. Two of 3 patients with SHH who did not receive salvage radiotherapy are survivors. CONCLUSION: A substantial proportion of young children who relapse following irradiation-sparing strategies can be salvaged. Neurocognitive and ototoxicity outcomes are being evaluated. Oxford University Press 2020-12-04 /pmc/articles/PMC7715107/ http://dx.doi.org/10.1093/neuonc/noaa222.500 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)
Erker, Craig
Larouche, Valérie
Margol, Ashley
Cacciotti, Chantel
Perreault, Sébastien
Cohen, Kenneth J
AbdelBaki, Mohamed S
Hukin, Juliette
Rassekh, Shahrad Rod
Eisenstat, David D
Wilson, Beverly
Knipstein, Jeffrey
Hoppmann, Anna L
Sandler, Eric S
Dorris, Kathleen
Fay-McClymont, Taryn B
Salloum, Ralph
Harrod, Virginia L
Crooks, Bruce
Finlay, Jonathan L
Bouffet, Eric
Lafay-Cousin, Lucie
MBCL-24. CAN YOUNG CHILDREN WITH RELAPSED MEDULLOBLASTOMA BE SALVAGED AFTER INITIAL IRRADIATION-SPARING APPROACHES?
title MBCL-24. CAN YOUNG CHILDREN WITH RELAPSED MEDULLOBLASTOMA BE SALVAGED AFTER INITIAL IRRADIATION-SPARING APPROACHES?
title_full MBCL-24. CAN YOUNG CHILDREN WITH RELAPSED MEDULLOBLASTOMA BE SALVAGED AFTER INITIAL IRRADIATION-SPARING APPROACHES?
title_fullStr MBCL-24. CAN YOUNG CHILDREN WITH RELAPSED MEDULLOBLASTOMA BE SALVAGED AFTER INITIAL IRRADIATION-SPARING APPROACHES?
title_full_unstemmed MBCL-24. CAN YOUNG CHILDREN WITH RELAPSED MEDULLOBLASTOMA BE SALVAGED AFTER INITIAL IRRADIATION-SPARING APPROACHES?
title_short MBCL-24. CAN YOUNG CHILDREN WITH RELAPSED MEDULLOBLASTOMA BE SALVAGED AFTER INITIAL IRRADIATION-SPARING APPROACHES?
title_sort mbcl-24. can young children with relapsed medulloblastoma be salvaged after initial irradiation-sparing approaches?
topic Medulloblastoma (Clinical)
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7715107/
http://dx.doi.org/10.1093/neuonc/noaa222.500
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