Department of Pediatric Neurosurgery, Children's Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102, China.
Department of Pediatric Neurosurgery, Children's Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102, China.
Clin Neurol Neurosurg. 2021 Jan;200:106345. doi: 10.1016/j.clineuro.2020.106345. Epub 2020 Nov 4.
Atypical choroid plexus papilloma (aCPP) is a newly introduced subtype of choroid plexus tumors (CPTs) defined by the World Health Organization (WHO) in 2007 and is characterized by intermediate characteristics between choroid plexus papilloma (CPP) and choroid plexus carcinoma (CPC). Currently, the available data describing the clinical features of aCPP in children are limited.
We performed a retrospective review of 24 pediatric patients with CPTs in our institute and focused on the clinical, radiological and histopathological features of 9 patients with aCPP.
The median age of aCPP patients was 12 (3-144) months, which was younger than the age of CPP patients (36 (5-132) months, P < 0.05). Of the 9 aCPPs, there were 4 cases of giant masses in the cerebral hemisphere, which was significantly higher than that in CPPs (44.4 % vs 0.0 %, P < 0.05). According to MRI analysis, cysts and necrosis (66.7 % vs 16.7 %, P < 0.05), peritumoral edema (55.6 % vs 8.3 %, P < 0.05) and blurred borders (55.6 % vs 8.3 %, P < 0.05) were more common in aCPPs than in CPPs. T1WI isointense signals were mainly observed in aCPPs and CPPs (aCPP66.7 % vs CPP58.3 %, P >0.05), while T2WI slightly low signals were more common in CPPs (CPP41.7 % vs aCPP0%, P < 0.05); moreover, the tumor volume of aCPPs was significantly larger than that of CPPs (aCPP78.3 cm3 vs 8.4 cm3, P < 0.05). For the DWI sequence scans, isointense signals were more common in aCPPs (aCPP77.8 %>CPP25.0 %, P < 0.05), while slightly low signals were more common in CPPs (CPP58.3 %>aCPP0%, P < 0.05). Both aCPPs and CPPs mainly showed homogeneously strong enhancement (aCPP66.7 % vs CPP91.7 %, P > 0.05). Interestingly, 1 aCPP showed annular enhancement. For the pathological and immunohistochemical studies, the Ki67 proliferation index was significantly higher in aCPPs than in CPPs (13 % vs 6%, P < 0.05), and the S-100(+)/Vim(+)/Syn(+) positive rate was higher in aCPPs (58.3 % vs 11.1 %, P < 0.05).
aCPP shows some distinctive clinical features compared with CPP, such as younger age, larger tumor size, more frequent necrosis and peritumoral edema, blurred borders, slightly low signals on T2WI and isointense signals on DWI, and a higher S-100(+)/Vim(+)/Syn(+) positive rate, which may provide more valuable evidence for differential diagnosis and clinical decisions surrounding aCPP.
非典型脉络丛乳头状瘤(aCPP)是 2007 年世界卫生组织(WHO)定义的脉络丛肿瘤(CPT)的一个新亚型,其特征是介于脉络丛乳头状瘤(CPP)和脉络丛癌(CPC)之间的中间特征。目前,描述儿童中 aCPP 临床特征的可用数据有限。
我们对我院 24 例 CPT 患儿进行回顾性研究,并重点分析 9 例 aCPP 患儿的临床、影像学和组织病理学特征。
aCPP 患儿的中位年龄为 12(3-144)个月,小于 CPP 患儿的年龄(36(5-132)个月,P < 0.05)。9 例 aCPP 中有 4 例为大脑半球的巨大肿块,明显高于 CPP(44.4%比 0.0%,P < 0.05)。根据 MRI 分析,aCPP 比 CPP 更常见囊变和坏死(66.7%比 16.7%,P < 0.05)、瘤周水肿(55.6%比 8.3%,P < 0.05)和边界模糊(55.6%比 8.3%,P < 0.05)。aCPP 和 CPP 主要表现为 T1WI 等信号(aCPP 66.7%比 CPP 58.3%,P > 0.05),而 T2WI 稍低信号更常见于 CPP(CPP 41.7%比 aCPP 0%,P < 0.05);此外,aCPP 的肿瘤体积明显大于 CPP(aCPP 78.3 cm3比 CPP 8.4 cm3,P < 0.05)。对于 DWI 序列扫描,aCPP 更常见等信号(aCPP 77.8%比 CPP 25.0%,P < 0.05),而 CPP 更常见稍低信号(CPP 58.3%比 aCPP 0%,P < 0.05)。aCPP 和 CPP 主要表现为均匀强化(aCPP 66.7%比 CPP 91.7%,P > 0.05)。有趣的是,1 例 aCPP 呈环形强化。在病理和免疫组织化学研究中,Ki67 增殖指数在 aCPP 中明显高于 CPP(13%比 6%,P < 0.05),S-100(+) / Vim(+) / Syn(+)阳性率在 aCPP 中较高(58.3%比 11.1%,P < 0.05)。
与 CPP 相比,aCPP 具有一些独特的临床特征,如年龄较小、肿瘤较大、更频繁的坏死和瘤周水肿、边界模糊、T2WI 稍低信号和 DWI 等信号,以及较高的 S-100(+) / Vim(+) / Syn(+)阳性率,这可能为 aCPP 的鉴别诊断和临床决策提供更有价值的证据。