Department of Pathology, The Children's Memorial Health Institute, Av. Dzieci Polskich 20, 04-730, Warsaw, Poland.
Department of Experimental and Clinical Neuropathology, Mossakowski Medical Research Centre Polish Academy of Sciences, A. Pawińskiego 5 Street, 02-106, Warsaw, Poland.
J Neurooncol. 2018 Jun;138(2):231-240. doi: 10.1007/s11060-018-2797-5. Epub 2018 Feb 9.
Medulloblastoma, the most common malignant pediatric brain tumor, is a heterogeneous disease, with the existence of at least four molecular types: Wingless (WNT), Sonic Hedgehog (SHH), Group 3 and Group 4 tumors. The latter two groups, which can be identified by an application of multi-gene expression or methylation profiling, show sometimes ambiguous categorization and are still classified for diagnostic reason as non-SHH/non-WNT medulloblastomas in updated WHO 2016 classification. In order to better characterize non-SHH/non-WNT tumors, we applied the method based on the Nanostring nCounter Technology, using the 26 genes codeset in 68 uniformly treated medulloblastoma patients. This allowed for identification of tumors, which shared common Group 3 and Group 4 gene signatures. We recognized three transcriptional groups within non-WNT/non-SHH tumors: Group 3, Group 4 and the Intermediate 3/4 Group. Group 3, in line with previously published results, showed poor prognosis with survival rate < 40%, frequent metastases, large cell/anaplastic pathology and presence of tumors with MYCC amplification. This is in contrast to patients from the Intermediate 3/4 Group who showed the best survival rate (100%). Overall and progression free survival were better for this group than for Group 3 (p = 0.001, for both) and Group 4 (p = 0.064 and p = 0.066, respectively). Our work supports the view that within the non-WNT/non-SHH tumors different risk groups exist and that the current two groups classifier may be not sufficient for proper clinical categorization of individual patients.
髓母细胞瘤是最常见的小儿脑恶性肿瘤,是一种异质性疾病,至少存在四种分子类型:无翅型(WNT)、Sonic Hedgehog(SHH)、Group 3 和 Group 4 肿瘤。后两种类型可通过多基因表达或甲基化谱分析来识别,有时分类不明确,在最新的 2016 年 WHO 分类中,仍被归类为非 SHH/非 WNT 髓母细胞瘤,用于诊断。为了更好地描述非 SHH/非 WNT 肿瘤,我们应用了基于 Nanostring nCounter 技术的方法,对 68 例经统一治疗的髓母细胞瘤患者的 26 个基因进行了分析。这使得我们能够识别具有共同 Group 3 和 Group 4 基因特征的肿瘤。在非 WNT/非 SHH 肿瘤中,我们发现了三个转录组:Group 3、Group 4 和 Intermediate 3/4 组。Group 3 与之前发表的结果一致,生存率<40%,常发生转移,大细胞/间变性病理和 MYCC 扩增的肿瘤。与 Intermediate 3/4 组的患者形成对比,后者的生存率最高(100%)。该组的总体生存率和无进展生存率均优于 Group 3(p=0.001,均)和 Group 4(p=0.064 和 p=0.066)。我们的工作支持以下观点:在非 WNT/非 SHH 肿瘤中存在不同的风险组,目前的两种分组方法可能不足以对个体患者进行适当的临床分类。