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

Autor:	Evans DG; Division of Evolution and Genomic Medicine, Manchester Centre for Genomic Medicine, Manchester Academic Health Science Centre, University of Manchester, St Mary's Hospital, Manchester Universities NHS Foundation Trust, Manchester, UK.; Geoffrey Jefferson Brain Research Centre, Manchester Academic Health Science Centre, Northern Care Alliance NHS Foundation Trust, University of Manchester, UK., Halliday D; Departments of Genomic Medicine, Neurology, Neurosurgery, ENT, Oxford University Hospitals NHS Foundation Trust., Obholzer R; Department of ENT, and Neurology, London, UK., Afridi S; Guy's and St Thomas' NHS Trust, London, UK., Forde C; Division of Evolution and Genomic Medicine, Manchester Centre for Genomic Medicine, Manchester Academic Health Science Centre, University of Manchester, St Mary's Hospital, Manchester Universities NHS Foundation Trust, Manchester, UK., Rutherford SA; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK., Hammerbeck-Ward C; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK., Lloyd SK; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK., Freeman SM; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK., Pathmanaban ON; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK.; Division of Neuroscience and Experimental Psychology, Faculty of Biology, Medicine and Health, University of Manchester, UK.; Geoffrey Jefferson Brain Research Centre, Manchester Academic Health Science Centre, Northern Care Alliance NHS Foundation Trust, University of Manchester, UK., Thomas OM; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK., Laitt RD; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK., Stivaros S; Division of Informatics, Imaging and Data Sciences, School of Health Sciences, Faculty of Biology, Medicine and Health, The University of Manchester, Manchester Academic Health Science Centre, Oxford Road, Manchester, M13 9PL, UK.; Academic Unit of Paediatric Radiology, Royal Manchester Children's Hospital, Central Manchester University Hospitals NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, UK.; Geoffrey Jefferson Brain Research Centre, Manchester Academic Health Science Centre, Northern Care Alliance NHS Foundation Trust, University of Manchester, UK., Kilday JP; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Manchester, UK.; Division of Cancer Sciences, Faculty of Biology, Medicine and Health, University of Manchester, UK., Vassallo G; Division of Evolution and Genomic Medicine, Manchester Centre for Genomic Medicine, Manchester Academic Health Science Centre, University of Manchester, St Mary's Hospital, Manchester Universities NHS Foundation Trust, Manchester, UK., McBain C; The Christie NHS Foundation Trust, Manchester, UK.; Geoffrey Jefferson Brain Research Centre, Manchester Academic Health Science Centre, Northern Care Alliance NHS Foundation Trust, University of Manchester, UK., Lavin T; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK., Paterson C; Division of Evolution and Genomic Medicine, Manchester Centre for Genomic Medicine, Manchester Academic Health Science Centre, University of Manchester, St Mary's Hospital, Manchester Universities NHS Foundation Trust, Manchester, UK., Whitfield G; Children's Brain Tumour Research Network (CBTRN), Royal Manchester Children's Hospital, Manchester, UK.; Division of Cancer Sciences, Faculty of Biology, Medicine and Health, University of Manchester, UK.; The Christie NHS Foundation Trust, Manchester, UK., McCabe MG; Division of Cancer Sciences, Faculty of Biology, Medicine and Health, University of Manchester, UK.; The Christie NHS Foundation Trust, Manchester, UK., Axon PR; Department of Otolaryngology, Cambridge University Hospitals NHS Foundation Trust, Cambridge, UK., Halliday J; Departments of Genomic Medicine, Neurology, Neurosurgery, ENT, Oxford University Hospitals NHS Foundation Trust., Mackeith S; Departments of Genomic Medicine, Neurology, Neurosurgery, ENT, Oxford University Hospitals NHS Foundation Trust., Parry A; Departments of Genomic Medicine, Neurology, Neurosurgery, ENT, Oxford University Hospitals NHS Foundation Trust., Harkness EF; Division of Informatics, Imaging and Data Sciences, School of Health Sciences, Faculty of Biology, Medicine and Health, The University of Manchester, Manchester Academic Health Science Centre, Oxford Road, Manchester, M13 9PL, UK.; Prevent Breast Cancer Centre, Wythenshawe Hospital, Manchester Universities NHS Foundation Trust, Manchester, UK., Buttimore J; Department of Otolaryngology, Cambridge University Hospitals NHS Foundation Trust, Cambridge, UK., King AT; Departments of Neurosurgery, Otolaryngology, Neuroradiology, and Neurology, Manchester Centre for Clinical Neurosciences Salford Royal Hospital, Northern Care Alliance NHS Foundation Trust, Manchester, UK.; Geoffrey Jefferson Brain Research Centre, Manchester Academic Health Science Centre, Northern Care Alliance NHS Foundation Trust, University of Manchester, UK.; Division of Cardiovascular Sciences, Faculty of Biology, Medicine and Health, University of Manchester, UK.
Jazyk:	angličtina
Zdroj:	Neuro-oncology advances [Neurooncol Adv] 2023 Mar 11; Vol. 5 (1), pp. vdad025. Date of Electronic Publication: 2023 Mar 11 (Print Publication: 2023).
DOI:	10.1093/noajnl/vdad025
Abstrakt:	Background: Radiation treatment of benign tumors in tumor predisposition syndromes is controversial, but short-term studies from treatment centers suggest 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 tumors. 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 in 1969. Kaplan-Meier analysis was performed for malignancy incidence and survival. Outcomes were central nervous system (CNS) M/MP (2cm annualized diameter growth) and survival from index tumor treatment. Results: In total, 1345 NF2 patients, 266 (133-Male) underwent radiation treatments between 1969 and 2021 with median first radiotherapy age of 32.9 (IQR = 22.4-46.0). Nine subsequent CNS malignancies/MPs were identified in cases with only 4 in 1079 untreated ( P < .001). Lifetime and 20-year CNS M/MP was ~6% in all irradiated patients-(4.9% for vestibular schwannomas [VS] radiotherapy) versus <1% in the non-irradiated population ( P < .001/.01). Controls were well matched for age at NF2 diagnosis and treatment (Males = 133%-50%) and had no M/MP in the CNS post-index tumor treatment ( P = .0016). Thirty-year survival from index tumor treatment was 45.62% (95% CI = 34.0-56.5) for cases and 66.4% (57.3-74.0) for controls ( P = .02), but was nonsignificantly worse for VS radiotherapy. Conclusion: NF2 patients should not be offered radiotherapy as first-line treatment of benign tumors and should be given a frank discussion of the potential 5% excess absolute risk of M/MP. Competing Interests: None other than many of the authors are surgeons, but 4 are oncologists. (© The Author(s) 2023. Published by Oxford University Press, the Society for Neuro-Oncology and the European Association of Neuro-Oncology.)
Databáze:	MEDLINE
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