Verbruggen Lisanne C, Kok Judith L, Teepen Jop C, Janssens Geert O, de Boer Charlotte M, Stalpers Lukas J A, Vernooij Meike W, van Dulmen-den Broeder Eline, Loonen Jacqueline J, van den Heuvel-Eibrink Marry M, Tissing Wim J E, van der Heiden-van der Loo Margriet, Versluys Anne Birgitta, Neggers Sebastian J C M M, van Leeuwen Flora E, Hoving Eelco W, Wesseling Pieter, Kremer Leontine C M, Ronckers Cécile M, van der Pal Helena J H
Princess Máxima Center for Pediatric Oncology, Utrecht, The Netherlands.
Princess Máxima Center for Pediatric Oncology, Utrecht, The Netherlands.
Eur J Cancer. 2021 Jun;150:240-249. doi: 10.1016/j.ejca.2021.03.021. Epub 2021 Apr 30.
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.
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.
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.
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.
脑膜瘤是儿童颅脑放疗（CrRT）后最常见的脑肿瘤。关于疾病进程以指导脑膜瘤临床管理的数据很少。本研究报告了荷兰儿童癌症幸存者（CCS）中经组织学确诊的脑膜瘤的临床特征。
荷兰儿童癌症长期效应（LATER）队列中的6015名CCS符合条件，其中1551名曾接受过CrRT。这些CCS在18岁之前（1963年至2002年之间）被诊断患有癌症，且未接受脑肿瘤筛查。我们通过与荷兰病理登记处（PALGA；1991 - 2018年）以及荷兰LATER登记处的记录链接，识别出经组织学确诊的脑膜瘤。我们从医疗记录中提取了有关诊断、治疗和随访的详细信息。
我们描述了93例患有脑膜瘤的CCS，其中89例（95.7%）接受了CrRT治疗（占1551例曾接受CrRT治疗者的5.7%；OR = 68）。诊断时的中位年龄为31.8岁（范围：13.2 - 50.5岁）。30名幸存者（32.3%）患有同步脑膜瘤；84例（90.3%）出现症状。仅16.1%的脑膜瘤在晚期效应诊所被检测到。随着时间推移，所有幸存者都接受了手术；三分之一的人还接受了放疗。在随访期间，38例（40.9%）幸存者出现了新的脑膜瘤，22例（23.7%）复发，至少4人因脑膜瘤死亡。
儿童癌症后经组织学确诊的脑膜瘤大多有症状时才被诊断出来，而非在晚期效应诊所的常规随访期间。这些脑膜瘤的发病年龄比偶然发现的脑膜瘤中位年轻20 - 25岁，常为多发，治疗后复发率高。告知CCS及其医疗保健提供者后续脑膜瘤的风险和症状至关重要。
