Wu Kuo-Sheng, Ho Donald Ming-Tak, Jou Shiann-Tarng, Yu Alice L, Tran Huy Minh, Liang Muh-Lii, Chen Hsin-Hung, Lee Yi-Yen, Chen Yi-Wei, Lin Shih-Chieh, Chang Feng-Chi, Tsai Min-Lan, Liu Yen-Lin, Lee Hsin-Lun, Hsieh Kevin Li-Chun, Huang Wen-Chang, Sung Shian-Ying, Chang Che-Chang, Changou Chun Austin, Liang Kung-Hao, Hsieh Tsung-Han, Liu Yun-Ru, Chao Meng-En, Chen Wan, Chu Shing-Shung, Cho Er-Chieh, Wong Tai-Tong
Graduate Institute of Clinical Medicine, College of Medicine, Taipei Medical University, Taipei 110, Taiwan.
Department of Pathology and Laboratory Medicine, Taipei Veterans General Hospital, Taipei 112, Taiwan.
Cancers (Basel). 2020 Mar 11;12(3):653. doi: 10.3390/cancers12030653.
In 2016, a project was initiated in Taiwan to adopt molecular diagnosis of childhood medulloblastoma (MB). In this study, we aimed to identify a molecular-clinical correlation and somatic mutation for exploring risk-adapted treatment, drug targets, and potential genetic predisposition. In total, 52 frozen tumor tissues of childhood MBs were collected. RNA sequencing (RNA-Seq) and DNA methylation array data were generated. Molecular subgrouping and clinical correlation analysis were performed. An adjusted Heidelberg risk stratification scheme was defined for updated clinical risk stratification. We selected 51 genes for somatic variant calling using RNA-Seq data. Relevant clinical findings were defined. Potential drug targets and genetic predispositions were explored. Four core molecular subgroups (WNT, SHH, Group 3, and Group 4) were identified. Genetic backgrounds of metastasis at diagnosis and extent of tumor resection were observed. The adjusted Heidelberg scheme showed its applicability. Potential drug targets were detected in the pathways of DNA damage response. Among the 10 patients with SHH MBs analyzed using whole exome sequencing studies, five patients exhibited potential genetic predispositions and four patients had relevant germline mutations. The findings of this study provide valuable information for updated risk adapted treatment and personalized care of childhood MBs in our cohort series and in Taiwan.
2016年,台湾启动了一个采用儿童髓母细胞瘤(MB)分子诊断的项目。在本研究中,我们旨在确定分子与临床的相关性以及体细胞突变,以探索风险适应性治疗、药物靶点和潜在的遗传易感性。总共收集了52例儿童MB的冷冻肿瘤组织。生成了RNA测序(RNA-Seq)和DNA甲基化阵列数据。进行了分子亚组分析和临床相关性分析。为更新临床风险分层定义了调整后的海德堡风险分层方案。我们使用RNA-Seq数据选择了51个基因进行体细胞变异检测。定义了相关的临床发现。探索了潜在的药物靶点和遗传易感性。确定了四个核心分子亚组(WNT、SHH、3组和4组)。观察到诊断时转移的遗传背景和肿瘤切除范围。调整后的海德堡方案显示了其适用性。在DNA损伤反应途径中检测到潜在的药物靶点。在使用全外显子测序研究分析的10例SHH MB患者中,5例患者表现出潜在的遗传易感性,4例患者有相关的种系突变。本研究的结果为我们队列系列和台湾地区儿童MB的更新风险适应性治疗和个性化护理提供了有价值的信息。