TY - JOUR
T1 - Clinical characteristics of subsequent histologically confirmed meningiomas in long-term childhood cancer survivors
T2 - A Dutch LATER study
AU - Verbruggen, Lisanne C.
AU - Kok, Judith L.
AU - Teepen, Jop C.
AU - Janssens, Geert O.
AU - de Boer, Charlotte M.
AU - Stalpers, Lukas J.A.
AU - Vernooij, Meike W.
AU - van Dulmen-den Broeder, Eline
AU - Loonen, Jacqueline J.
AU - van den Heuvel-Eibrink, Marry M.
AU - Tissing, Wim J.E.
AU - van der Heiden-van der Loo, Margriet
AU - Versluys, Anne Birgitta
AU - Neggers, Sebastian J.C.M.M.
AU - van Leeuwen, Flora E.
AU - Hoving, Eelco W.
AU - Wesseling, Pieter
AU - Kremer, Leontine C.M.
AU - Ronckers, Cécile M.
AU - van der Pal, Helena J.H.
AU - Louwerens, Marloes
AU - de Vries, Andrica
AU - Jaspers, Monique
AU - Hollema, Nynke
AU - den Hartogh, Jaap
AU - Schouten-van Meeteren, Netteke
N1 - Funding Information:
This work was supported by KiKa Children Cancer Free Foundation (grant number 246 title: Radiotherapy-related meningiomas, cerebrovascular events, and cataract in childhood cancer survivors: a DCOG-LATER project).
Publisher Copyright:
© 2021 Elsevier Ltd
Copyright © 2021 Elsevier Ltd. All rights reserved.
PY - 2021/6
Y1 - 2021/6
N2 - BACKGROUND: Meningiomas are the most frequent brain tumours occurring after pediatric cranial radiotherapy (CrRT). Data on course of disease, to inform clinical management of meningiomas, are sparse. This study reports the clinical characteristics of histologically confirmed meningiomas in childhood cancer survivors (CCS) in the Netherlands.METHODS: In total, 6015 CCS from the Dutch Long-Term Effects After Childhood Cancer (LATER) cohort were eligible, including 1551 with prior CrRT. These CCS were diagnosed with cancer age <18 y (between 1963 and 2002) and are not subject to brain tumour screening. We identified histologically confirmed meningiomas by record linkage with the Dutch Pathology Registry (PALGA; 1991-2018), and in the Dutch LATER registry. We extracted details regarding diagnosis, treatment, and follow-up from medical records.RESULTS: We described 93 CCS with meningioma, of whom 89 (95.7%) were treated with CrRT (5.7% of 1551 with prior CrRT; OR = 68). Median age at diagnosis was 31.8 y (range: 13.2-50.5). Thirty survivors (32.3%) had synchronous meningiomas; 84 (90.3%) presented with symptoms. Only 16.1% of meningioma was detected at late effects clinics. Over time, all survivors had surgery; one-third also received radiotherapy. During follow-up 38 (40.9%), survivors developed new meningiomas, 22(23.7%) recurrences and at least four died due to the meningioma.CONCLUSIONS: Histologically confirmed meningiomas after childhood cancer are mostly diagnosed with symptoms and not during routine follow-up at late effects clinics. The meningiomas occur at a median of 20-25 y younger age than incidental meningiomas, are frequently multiple and recurrence after treatment is high. It is crucial to inform CCS and healthcare providers about risk and symptoms of subsequent meningiomas.
AB - BACKGROUND: Meningiomas are the most frequent brain tumours occurring after pediatric cranial radiotherapy (CrRT). Data on course of disease, to inform clinical management of meningiomas, are sparse. This study reports the clinical characteristics of histologically confirmed meningiomas in childhood cancer survivors (CCS) in the Netherlands.METHODS: In total, 6015 CCS from the Dutch Long-Term Effects After Childhood Cancer (LATER) cohort were eligible, including 1551 with prior CrRT. These CCS were diagnosed with cancer age <18 y (between 1963 and 2002) and are not subject to brain tumour screening. We identified histologically confirmed meningiomas by record linkage with the Dutch Pathology Registry (PALGA; 1991-2018), and in the Dutch LATER registry. We extracted details regarding diagnosis, treatment, and follow-up from medical records.RESULTS: We described 93 CCS with meningioma, of whom 89 (95.7%) were treated with CrRT (5.7% of 1551 with prior CrRT; OR = 68). Median age at diagnosis was 31.8 y (range: 13.2-50.5). Thirty survivors (32.3%) had synchronous meningiomas; 84 (90.3%) presented with symptoms. Only 16.1% of meningioma was detected at late effects clinics. Over time, all survivors had surgery; one-third also received radiotherapy. During follow-up 38 (40.9%), survivors developed new meningiomas, 22(23.7%) recurrences and at least four died due to the meningioma.CONCLUSIONS: Histologically confirmed meningiomas after childhood cancer are mostly diagnosed with symptoms and not during routine follow-up at late effects clinics. The meningiomas occur at a median of 20-25 y younger age than incidental meningiomas, are frequently multiple and recurrence after treatment is high. It is crucial to inform CCS and healthcare providers about risk and symptoms of subsequent meningiomas.
KW - Adolescent
KW - Adult
KW - Age of Onset
KW - Cancer Survivors
KW - Female
KW - Humans
KW - Male
KW - Meningeal Neoplasms/mortality
KW - Meningioma/epidemiology
KW - Middle Aged
KW - Neoplasms, Multiple Primary/mortality
KW - Neoplasms, Second Primary/mortality
KW - Netherlands/epidemiology
KW - Retrospective Studies
KW - Risk Assessment
KW - Risk Factors
KW - Time Factors
KW - Treatment Outcome
KW - Young Adult
KW - Cranial radiotherapy
KW - Subsequent meningioma
KW - Childhood adolescent young adult (CAYA) cancer survivors
UR - http://www.scopus.com/inward/record.url?scp=85104914764&partnerID=8YFLogxK
U2 - 10.1016/j.ejca.2021.03.021
DO - 10.1016/j.ejca.2021.03.021
M3 - Article
C2 - 33934061
AN - SCOPUS:85104914764
SN - 0959-8049
VL - 150
SP - 240
EP - 249
JO - European Journal of Cancer
JF - European Journal of Cancer
ER -