The effectiveness and safety of proton beam radiation therapy in children and young adults with Central Nervous System (CNS) tumours: a systematic review

Jayne S Wilson; Caroline Main; Nicky Thorp; Roger E Taylor; Saimma Majothi; Pamela R Kearns; Martin English; Madhumita Dandapani; Robert Phillips; Keith Wheatley; Barry Pizer

doi:10.1007/s11060-023-04510-4

The effectiveness and safety of proton beam radiation therapy in children and young adults with Central Nervous System (CNS) tumours: a systematic review

Jayne S Wilson, Caroline Main, Nicky Thorp, Roger E Taylor, Saimma Majothi, Pamela R Kearns, Martin English, Madhumita Dandapani, Robert Phillips, Keith Wheatley, Barry Pizer

Research output: Contribution to journal › Review article › peer-review

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Abstract

BACKGROUND: Central nervous system (CNS) tumours account for around 25% of childhood neoplasms. With multi-modal therapy, 5-year survival is at around 75% in the UK. Conventional photon radiotherapy has made significant contributions to survival, but can be associated with long-term side effects. Proton beam radiotherapy (PBT) reduces the volume of irradiated tissue outside the tumour target volume which may potentially reduce toxicity. Our aim was to assess the effectiveness and safety of PBT and make recommendations for future research for this evolving treatment.

METHODS: A systematic review assessing the effects of PBT for treating CNS tumours in children/young adults was undertaken using methods recommended by Cochrane and reported using PRISMA guidelines. Any study design was included where clinical and toxicity outcomes were reported. Searches were to May 2021, with a narrative synthesis employed.

RESULTS: Thirty-one case series studies involving 1731 patients from 10 PBT centres were included. Eleven studies involved children with medulloblastoma / primitive neuroectodermal tumours (n = 712), five ependymoma (n = 398), four atypical teratoid/rhabdoid tumour (n = 72), six craniopharyngioma (n = 272), three low-grade gliomas (n = 233), one germ cell tumours (n = 22) and one pineoblastoma (n = 22). Clinical outcomes were the most frequently reported with overall survival values ranging from 100 to 28% depending on the tumour type. Endocrine outcomes were the most frequently reported toxicity outcomes with quality of life the least reported.

CONCLUSIONS: This review highlights areas of uncertainty in this research area. A well-defined, well-funded research agenda is needed to best maximise the potential of PBT.

SYSTEMATIC REVIEW REGISTRATION: PROSPERO-CRD42016036802.

Original language	English
Number of pages	34
Journal	Journal of Neuro-Oncology
Early online date	31 Jan 2024
DOIs	https://doi.org/10.1007/s11060-023-04510-4
Publication status	E-pub ahead of print - 31 Jan 2024

Bibliographical note

This paper presents independent research funded by the National Institute for Health Research (NIHR) under its Research for Patient Benefit (RfPB) Programme (Grant Reference Number PB-PG-1112–29122). The views expressed are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health.

Keywords

Children
CNS tumours
Proton beam radiotherapy
Brain
Photon beam
Systematic review

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WilsonJ2024EffectivenessFinal published version, 1.13 MBLicence: Creative Commons: Attribution (CC BY)

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Wilson, J. S., Main, C., Thorp, N., Taylor, R. E., Majothi, S., Kearns, P. R., English, M., Dandapani, M., Phillips, R., Wheatley, K., & Pizer, B. (2024). The effectiveness and safety of proton beam radiation therapy in children and young adults with Central Nervous System (CNS) tumours: a systematic review. Journal of Neuro-Oncology. Advance online publication. https://doi.org/10.1007/s11060-023-04510-4

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abstract = "BACKGROUND: Central nervous system (CNS) tumours account for around 25% of childhood neoplasms. With multi-modal therapy, 5-year survival is at around 75% in the UK. Conventional photon radiotherapy has made significant contributions to survival, but can be associated with long-term side effects. Proton beam radiotherapy (PBT) reduces the volume of irradiated tissue outside the tumour target volume which may potentially reduce toxicity. Our aim was to assess the effectiveness and safety of PBT and make recommendations for future research for this evolving treatment.METHODS: A systematic review assessing the effects of PBT for treating CNS tumours in children/young adults was undertaken using methods recommended by Cochrane and reported using PRISMA guidelines. Any study design was included where clinical and toxicity outcomes were reported. Searches were to May 2021, with a narrative synthesis employed.RESULTS: Thirty-one case series studies involving 1731 patients from 10 PBT centres were included. Eleven studies involved children with medulloblastoma / primitive neuroectodermal tumours (n = 712), five ependymoma (n = 398), four atypical teratoid/rhabdoid tumour (n = 72), six craniopharyngioma (n = 272), three low-grade gliomas (n = 233), one germ cell tumours (n = 22) and one pineoblastoma (n = 22). Clinical outcomes were the most frequently reported with overall survival values ranging from 100 to 28% depending on the tumour type. Endocrine outcomes were the most frequently reported toxicity outcomes with quality of life the least reported.CONCLUSIONS: This review highlights areas of uncertainty in this research area. A well-defined, well-funded research agenda is needed to best maximise the potential of PBT.SYSTEMATIC REVIEW REGISTRATION: PROSPERO-CRD42016036802.",

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note = "This paper presents independent research funded by the National Institute for Health Research (NIHR) under its Research for Patient Benefit (RfPB) Programme (Grant Reference Number PB-PG-1112–29122). The views expressed are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health.",

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T1 - The effectiveness and safety of proton beam radiation therapy in children and young adults with Central Nervous System (CNS) tumours

T2 - a systematic review

AU - Wilson, Jayne S

AU - Main, Caroline

AU - Thorp, Nicky

AU - Taylor, Roger E

AU - Majothi, Saimma

AU - Kearns, Pamela R

AU - English, Martin

AU - Dandapani, Madhumita

AU - Phillips, Robert

AU - Wheatley, Keith

AU - Pizer, Barry

N1 - This paper presents independent research funded by the National Institute for Health Research (NIHR) under its Research for Patient Benefit (RfPB) Programme (Grant Reference Number PB-PG-1112–29122). The views expressed are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health.

PY - 2024/1/31

Y1 - 2024/1/31

N2 - BACKGROUND: Central nervous system (CNS) tumours account for around 25% of childhood neoplasms. With multi-modal therapy, 5-year survival is at around 75% in the UK. Conventional photon radiotherapy has made significant contributions to survival, but can be associated with long-term side effects. Proton beam radiotherapy (PBT) reduces the volume of irradiated tissue outside the tumour target volume which may potentially reduce toxicity. Our aim was to assess the effectiveness and safety of PBT and make recommendations for future research for this evolving treatment.METHODS: A systematic review assessing the effects of PBT for treating CNS tumours in children/young adults was undertaken using methods recommended by Cochrane and reported using PRISMA guidelines. Any study design was included where clinical and toxicity outcomes were reported. Searches were to May 2021, with a narrative synthesis employed.RESULTS: Thirty-one case series studies involving 1731 patients from 10 PBT centres were included. Eleven studies involved children with medulloblastoma / primitive neuroectodermal tumours (n = 712), five ependymoma (n = 398), four atypical teratoid/rhabdoid tumour (n = 72), six craniopharyngioma (n = 272), three low-grade gliomas (n = 233), one germ cell tumours (n = 22) and one pineoblastoma (n = 22). Clinical outcomes were the most frequently reported with overall survival values ranging from 100 to 28% depending on the tumour type. Endocrine outcomes were the most frequently reported toxicity outcomes with quality of life the least reported.CONCLUSIONS: This review highlights areas of uncertainty in this research area. A well-defined, well-funded research agenda is needed to best maximise the potential of PBT.SYSTEMATIC REVIEW REGISTRATION: PROSPERO-CRD42016036802.

AB - BACKGROUND: Central nervous system (CNS) tumours account for around 25% of childhood neoplasms. With multi-modal therapy, 5-year survival is at around 75% in the UK. Conventional photon radiotherapy has made significant contributions to survival, but can be associated with long-term side effects. Proton beam radiotherapy (PBT) reduces the volume of irradiated tissue outside the tumour target volume which may potentially reduce toxicity. Our aim was to assess the effectiveness and safety of PBT and make recommendations for future research for this evolving treatment.METHODS: A systematic review assessing the effects of PBT for treating CNS tumours in children/young adults was undertaken using methods recommended by Cochrane and reported using PRISMA guidelines. Any study design was included where clinical and toxicity outcomes were reported. Searches were to May 2021, with a narrative synthesis employed.RESULTS: Thirty-one case series studies involving 1731 patients from 10 PBT centres were included. Eleven studies involved children with medulloblastoma / primitive neuroectodermal tumours (n = 712), five ependymoma (n = 398), four atypical teratoid/rhabdoid tumour (n = 72), six craniopharyngioma (n = 272), three low-grade gliomas (n = 233), one germ cell tumours (n = 22) and one pineoblastoma (n = 22). Clinical outcomes were the most frequently reported with overall survival values ranging from 100 to 28% depending on the tumour type. Endocrine outcomes were the most frequently reported toxicity outcomes with quality of life the least reported.CONCLUSIONS: This review highlights areas of uncertainty in this research area. A well-defined, well-funded research agenda is needed to best maximise the potential of PBT.SYSTEMATIC REVIEW REGISTRATION: PROSPERO-CRD42016036802.

KW - Children

KW - CNS tumours

KW - Proton beam radiotherapy

KW - Brain

KW - Photon beam

KW - Systematic review

U2 - 10.1007/s11060-023-04510-4

DO - 10.1007/s11060-023-04510-4

M3 - Review article

C2 - 38294638

SN - 0167-594X

JO - Journal of Neuro-Oncology

JF - Journal of Neuro-Oncology

ER -

The effectiveness and safety of proton beam radiation therapy in children and young adults with Central Nervous System (CNS) tumours: a systematic review

Abstract

Bibliographical note

Keywords

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