Radiation treatment of benign tumours in NF2-related-schwannomatosis: a national study of 266 irradiated patients showing a significant increase in malignancy/malignant progression

D Gareth Evans; Dorothy Halliday; Rupert Obholzer; Shazia Afridi; Claire Forde; Scott A Rutherford; Charlotte Hammerbeck-Ward; Simon K. Lloyd; Simon M. Freeman; Omar Pathmanaban; Owen  Thomas; Roger D. Laitt; Stavros Stivaros; John-Paul Kilday; Grace Vasallo; Catherine Mcbain; Timothy Lavin; Chay Paterson; Gillian Whitfield; Patrick R. Axon; Martin Mccabe; Jane Halliday; Samuel  MacKeith; Allyson Parry; English Specialist NF2 Research Group; Elaine Harkness; Juliette Buttimore; Andrew King

Radiation treatment of benign tumours in NF2-related-schwannomatosis: a national study of 266 irradiated patients showing a significant increase in malignancy/malignant progression

D Gareth Evans, Dorothy Halliday, Rupert Obholzer, Shazia Afridi, Claire Forde, Scott A Rutherford, Charlotte Hammerbeck-Ward, Simon K. Lloyd, Simon M. Freeman, Omar Pathmanaban, Owen Thomas, Roger D. Laitt, Stavros Stivaros, John-Paul Kilday, Grace Vasallo, Catherine Mcbain, Timothy Lavin, Chay Paterson, Gillian Whitfield, Patrick R. AxonMartin Mccabe, Jane Halliday, Samuel MacKeith, Allyson Parry, English Specialist NF2 Research Group, Elaine Harkness, Juliette Buttimore, Andrew King

Research output: Contribution to journal › Article › peer-review

Abstract

Background: Radiation treatment of benign tumours in tumour-predisposition syndromes is controversial, but short-term studies from treatment centres suggests safety despite apparent radiation associated malignancy being reported. We determined whether radiation treatment in NF2-related-schwannomatosis patients is associated with increased rates of subsequent malignancy-(M)/malignant progression-(MP).
Methods: All UK patients with NF2 were eligible if they had a clinical/molecular diagnosis. Cases were NF2 patients treated with radiation for benign tumours. Controls were matched for treatment location with surgical/medical treatments based on age and year of treatment. Prospective data collection began in 1990 with addition of retrospective cases to 1969. Kaplan-Meier analysis was performed for malignancy incidence and survival. Outcomes were CNS M/MP (2cm annualised diameter growth) and survival from index tumour treatment.
Results: 1345 NF2 patients, 266 (133-Male) underwent radiation treatments between 1969-2021 with median first radiotherapy aged 32.9-(IQR=22.4-46.0). Nine subsequent CNS malignancies/malignant progressions were identified in cases with only four in 1079 untreated-(pConclusion: -NF2 patients should not be offered radiotherapy as first line treatment of benign tumours and should be given a frank discussion of the potential 5% excess absolute risk of M/MP.

Original language	English
Journal	Neuro-Oncology Advances
Publication status	Accepted/In press - 5 Mar 2023

Keywords

NF2
schwannoma
MPNST
malignant transformation
radiotherapy

Research Beacons, Institutes and Platforms

Manchester Cancer Research Centre

NIHR Manchester Biomedical Research Centre
Bruce, I., Lord, G., Lennon, R., Black, G., Wedge, D., Morris, A., Hussell, T., Sharrocks, A., Stivaros, S., Buch, M., Gough, J., Kostarelos, K., Thistlethwaite, F., Kadler, K., Barton, A., Hyrich, K., Mcbeth, J., O'Neill, T., Vestbo, J., Simpson, A., Singh, S., Smith, J., Felton, T., Murray, C., Griffiths, C., Cullum, N., Rhodes, L., Warren, R., Paus, R., Dumville, J., Viros Usandizaga, A., Keavney, B., Tomaszewski, M., Allan, S., Body, R., Cartwright, E., Heagerty, A., Kalra, P., Miller, C., Rutter, M., Smith, C., Trafford, A., Evans, D., Crosbie, E., Crosbie, P., Harvie, M., Howell, S., Renehan, A., Dive, C., Blackhall, F., Landers, D., Krebs, M., Cook, N., Clarke, R., Taylor, S., Jorgensen, C., Lorigan, P., Jayson, G., Valle, J., Mccabe, M., Armstrong, A., Freitas, A., Illidge, T., Choudhury, A., Hoskin, P., West, C., Van Herk, M., Faivre-Finn, C., Bristow, R., Kirkby, K., Birtle, A., Mackay, R., Radford, J., Linton, K., Higham, C., Munro, K., Plack, C., Arden Armitage, C., Bruce, I., Moore, D., Saunders, G., Stone, M., Haddock, G., Lewis, S., Elliott, R., Green, J., Lovell, K., Morrison, A., Shaw, J., Bucci, S., Ainsworth, J., Webb, R., Newman, W., Banka, S., Clayton-Smith, J., Payne, K., Moldovan, R., Wynn, R. & Jones, S.
1/12/22 → 30/11/27
Project: Research

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Evans, D. G., Halliday, D., Obholzer, R., Afridi, S., Forde, C., Rutherford, S. A., Hammerbeck-Ward, C., Lloyd, S. K., Freeman, S. M., Pathmanaban, O., Thomas, O., Laitt, R. D., Stivaros, S., Kilday, J-P., Vasallo, G., Mcbain, C., Lavin, T., Paterson, C., Whitfield, G., ... King, A. (Accepted/In press). Radiation treatment of benign tumours in NF2-related-schwannomatosis: a national study of 266 irradiated patients showing a significant increase in malignancy/malignant progression. Neuro-Oncology Advances.

@article{a3dcf1520a0d4ebfa85bf4e67db9b67f,

title = "Radiation treatment of benign tumours in NF2-related-schwannomatosis: a national study of 266 irradiated patients showing a significant increase in malignancy/malignant progression",

abstract = "Background: Radiation treatment of benign tumours in tumour-predisposition syndromes is controversial, but short-term studies from treatment centres suggests safety despite apparent radiation associated malignancy being reported. We determined whether radiation treatment in NF2-related-schwannomatosis patients is associated with increased rates of subsequent malignancy-(M)/malignant progression-(MP). Methods: All UK patients with NF2 were eligible if they had a clinical/molecular diagnosis. Cases were NF2 patients treated with radiation for benign tumours. Controls were matched for treatment location with surgical/medical treatments based on age and year of treatment. Prospective data collection began in 1990 with addition of retrospective cases to 1969. Kaplan-Meier analysis was performed for malignancy incidence and survival. Outcomes were CNS M/MP (2cm annualised diameter growth) and survival from index tumour treatment. Results: 1345 NF2 patients, 266 (133-Male) underwent radiation treatments between 1969-2021 with median first radiotherapy aged 32.9-(IQR=22.4-46.0). Nine subsequent CNS malignancies/malignant progressions were identified in cases with only four in 1079 untreated-(pConclusion: -NF2 patients should not be offered radiotherapy as first line treatment of benign tumours and should be given a frank discussion of the potential 5% excess absolute risk of M/MP. ",

keywords = "NF2, schwannoma, MPNST, malignant transformation, radiotherapy",

author = "Evans, {D Gareth} and Dorothy Halliday and Rupert Obholzer and Shazia Afridi and Claire Forde and Rutherford, {Scott A} and Charlotte Hammerbeck-Ward and Lloyd, {Simon K.} and Freeman, {Simon M.} and Omar Pathmanaban and Owen Thomas and Laitt, {Roger D.} and Stavros Stivaros and John-Paul Kilday and Grace Vasallo and Catherine Mcbain and Timothy Lavin and Chay Paterson and Gillian Whitfield and Axon, {Patrick R.} and Martin Mccabe and Jane Halliday and Samuel MacKeith and Allyson Parry and {English Specialist NF2 Research Group} and Elaine Harkness and Juliette Buttimore and Andrew King",

year = "2023",

month = mar,

day = "5",

language = "English",

journal = "Neuro-Oncology Advances",

issn = "2632-2498",

publisher = "Oxford University Press",

}

Evans, DG, Halliday, D, Obholzer, R, Afridi, S, Forde, C, Rutherford, SA, Hammerbeck-Ward, C, Lloyd, SK, Freeman, SM, Pathmanaban, O, Thomas, O, Laitt, RD, Stivaros, S, Kilday, J-P, Vasallo, G, Mcbain, C, Lavin, T, Paterson, C, Whitfield, G, Axon, PR, Mccabe, M, Halliday, J, MacKeith, S, Parry, A, English Specialist NF2 Research Group, Harkness, E, Buttimore, J & King, A 2023, 'Radiation treatment of benign tumours in NF2-related-schwannomatosis: a national study of 266 irradiated patients showing a significant increase in malignancy/malignant progression', Neuro-Oncology Advances.

TY - JOUR

T1 - Radiation treatment of benign tumours in NF2-related-schwannomatosis: a national study of 266 irradiated patients showing a significant increase in malignancy/malignant progression

AU - Evans, D Gareth

AU - Halliday, Dorothy

AU - Obholzer, Rupert

AU - Afridi, Shazia

AU - Forde, Claire

AU - Rutherford, Scott A

AU - Hammerbeck-Ward, Charlotte

AU - Lloyd, Simon K.

AU - Freeman, Simon M.

AU - Pathmanaban, Omar

AU - Thomas, Owen

AU - Laitt, Roger D.

AU - Stivaros, Stavros

AU - Kilday, John-Paul

AU - Vasallo, Grace

AU - Mcbain, Catherine

AU - Lavin, Timothy

AU - Paterson, Chay

AU - Whitfield, Gillian

AU - Axon, Patrick R.

AU - Mccabe, Martin

AU - Halliday, Jane

AU - MacKeith, Samuel

AU - Parry, Allyson

AU - English Specialist NF2 Research Group

AU - Harkness, Elaine

AU - Buttimore, Juliette

AU - King, Andrew

PY - 2023/3/5

Y1 - 2023/3/5

N2 - Background: Radiation treatment of benign tumours in tumour-predisposition syndromes is controversial, but short-term studies from treatment centres suggests safety despite apparent radiation associated malignancy being reported. We determined whether radiation treatment in NF2-related-schwannomatosis patients is associated with increased rates of subsequent malignancy-(M)/malignant progression-(MP). Methods: All UK patients with NF2 were eligible if they had a clinical/molecular diagnosis. Cases were NF2 patients treated with radiation for benign tumours. Controls were matched for treatment location with surgical/medical treatments based on age and year of treatment. Prospective data collection began in 1990 with addition of retrospective cases to 1969. Kaplan-Meier analysis was performed for malignancy incidence and survival. Outcomes were CNS M/MP (2cm annualised diameter growth) and survival from index tumour treatment. Results: 1345 NF2 patients, 266 (133-Male) underwent radiation treatments between 1969-2021 with median first radiotherapy aged 32.9-(IQR=22.4-46.0). Nine subsequent CNS malignancies/malignant progressions were identified in cases with only four in 1079 untreated-(pConclusion: -NF2 patients should not be offered radiotherapy as first line treatment of benign tumours and should be given a frank discussion of the potential 5% excess absolute risk of M/MP.

AB - Background: Radiation treatment of benign tumours in tumour-predisposition syndromes is controversial, but short-term studies from treatment centres suggests safety despite apparent radiation associated malignancy being reported. We determined whether radiation treatment in NF2-related-schwannomatosis patients is associated with increased rates of subsequent malignancy-(M)/malignant progression-(MP). Methods: All UK patients with NF2 were eligible if they had a clinical/molecular diagnosis. Cases were NF2 patients treated with radiation for benign tumours. Controls were matched for treatment location with surgical/medical treatments based on age and year of treatment. Prospective data collection began in 1990 with addition of retrospective cases to 1969. Kaplan-Meier analysis was performed for malignancy incidence and survival. Outcomes were CNS M/MP (2cm annualised diameter growth) and survival from index tumour treatment. Results: 1345 NF2 patients, 266 (133-Male) underwent radiation treatments between 1969-2021 with median first radiotherapy aged 32.9-(IQR=22.4-46.0). Nine subsequent CNS malignancies/malignant progressions were identified in cases with only four in 1079 untreated-(pConclusion: -NF2 patients should not be offered radiotherapy as first line treatment of benign tumours and should be given a frank discussion of the potential 5% excess absolute risk of M/MP.

KW - NF2

KW - schwannoma

KW - MPNST

KW - malignant transformation

KW - radiotherapy

M3 - Article

SN - 2632-2498

JO - Neuro-Oncology Advances

JF - Neuro-Oncology Advances

ER -

Radiation treatment of benign tumours in NF2-related-schwannomatosis: a national study of 266 irradiated patients showing a significant increase in malignancy/malignant progression

Abstract

Keywords

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