Department of Paediatric Oncology and Haematology, Faculty of Medicine, Jagiellonian University, Medical College, Krakow, Poland.
Department of Paediatrics, Haematology and Oncology, Medical University of Gdansk, Gdansk, Poland.
BMC Cancer. 2022 Jun 25;22(1):701. doi: 10.1186/s12885-022-09776-x.
Although isolated central nervous system (CNS) relapses are rare, they may become a serious clinical problem in intensively treated patients with high-risk neuroblastoma (NBL). The aim of this study is the presentation and assessment of the incidence and clinical course of isolated CNS relapses. Retrospective analysis involved 848 NBL patients treated from 2001 to 2019 at 8 centres of the Polish Paediatric Solid Tumours Study Group (PPSTSG). Group characteristics at diagnosis, treatment and patterns of relapse were analysed. Observation was completed in December 2020. We analysed 286 high risk patients, including 16 infants. Isolated CNS relapse, defined as the presence of a tumour in brain parenchyma or leptomeningeal involvement, was found in 13 patients (4.5%; 8.4% of all relapses), all of whom were stage 4 at diagnosis. Isolated CNS relapses seem to be more common in young patients with stage 4 MYCN amplified NBL, and in this group they may occur early during first line therapy. The only or the first symptom may be bleeding into the CNS, especially in younger children, even without a clear relapse picture on imaging, or the relapse may be clinically asymptomatic and found during routine screening. Although the incidence of isolated CNS relapses is not statistically significantly higher in patients after immunotherapy, their occurrence should be carefully monitored, especially in intensively treated infants, with potential disruption of the brain-blood barrier.
尽管孤立性中枢神经系统(CNS)复发较为罕见,但在高危神经母细胞瘤(NBL)的强化治疗患者中,它们可能成为严重的临床问题。本研究旨在介绍和评估孤立性 CNS 复发的发生率和临床病程。回顾性分析涉及波兰儿科实体瘤研究组(PPSTSG)的 8 个中心在 2001 年至 2019 年期间治疗的 848 名 NBL 患者。分析了诊断、治疗和复发模式的组特征。观察于 2020 年 12 月完成。我们分析了 286 名高危患者,包括 16 名婴儿。孤立性 CNS 复发定义为脑实质肿瘤或软脑膜受累,在 13 名患者(4.5%;所有复发的 8.4%)中发现,所有患者均为诊断时的 4 期。孤立性 CNS 复发似乎在 4 期 MYCN 扩增 NBL 的年轻患者中更为常见,在该组中,它们可能在一线治疗早期发生。唯一或首发症状可能是 CNS 出血,尤其是年龄较小的儿童,即使在成像上没有明确的复发迹象,或者复发可能是临床无症状的,在常规筛查中发现。尽管免疫治疗后孤立性 CNS 复发的发生率在统计学上没有显著增加,但应仔细监测其发生,尤其是在强化治疗的婴儿中,因为他们的血脑屏障可能受到破坏。