Wang Yongfeng, Wang Long, Guan Shan, Cao Wenming, Wang Hao, Chen Zhenghu, Zhao Yanling, Yu Yang, Zhang Huiyuan, Pang Jonathan C, Huang Sophia L, Akiyama Yo, Yang Yifan, Sun Wenjing, Xu Xin, Shi Yan, Zhang Hong, Kim Eugene S, Muscal Jodi A, Lu Fengmin, Yang Jianhua
Department of Microbiology, Peking University Health Science Center, Beijing 100191, China.
Texas Children's Cancer Center, Department of Pediatrics, Dan L. Duncan Cancer Center, Baylor College of Medicine, Houston, Texas 77030, USA.
Sci Rep. 2016 Jan 20;6:19423. doi: 10.1038/srep19423.
ALK receptor tyrosine kinase has been shown to be a therapeutic target in neuroblastoma. Germline ALK activating mutations are responsible for the majority of hereditary neuroblastoma and somatic ALK activating mutations are also frequently observed in sporadic cases of advanced NB. Crizotinib, a first-line therapy in the treatment of advanced non-small cell lung cancer (NSCLC) harboring ALK rearrangements, demonstrates striking efficacy against ALK-rearranged NB. However, crizotinib fails to effectively inhibit the activity of ALK when activating mutations are present within its kinase domain, as with the F1174L mutation. Here we show that a new ALK inhibitor AZD3463 effectively suppressed the proliferation of NB cell lines with wild type ALK (WT) as well as ALK activating mutations (F1174L and D1091N) by blocking the ALK-mediated PI3K/AKT/mTOR pathway and ultimately induced apoptosis and autophagy. In addition, AZD3463 enhanced the cytotoxic effects of doxorubicin on NB cells. AZD3463 also exhibited significant therapeutic efficacy on the growth of the NB tumors with WT and F1174L activating mutation ALK in orthotopic xenograft mouse models. These results indicate that AZD3463 is a promising therapeutic agent in the treatment of NB.
间变性淋巴瘤激酶(ALK)受体酪氨酸激酶已被证明是神经母细胞瘤的一个治疗靶点。胚系ALK激活突变是大多数遗传性神经母细胞瘤的病因,而体细胞ALK激活突变在晚期神经母细胞瘤(NB)的散发病例中也经常被观察到。克唑替尼是治疗携带ALK重排的晚期非小细胞肺癌(NSCLC)的一线疗法,对ALK重排的NB显示出显著疗效。然而,当ALK激酶结构域内存在激活突变时,如F1174L突变,克唑替尼无法有效抑制ALK的活性。在此我们表明,一种新的ALK抑制剂AZD3463通过阻断ALK介导的PI3K/AKT/mTOR途径,有效抑制了具有野生型ALK(WT)以及ALK激活突变(F1174L和D1091N)的NB细胞系的增殖,并最终诱导细胞凋亡和自噬。此外,AZD3463增强了阿霉素对NB细胞的细胞毒性作用。在原位异种移植小鼠模型中,AZD3463对具有WT和F1174L激活突变ALK的NB肿瘤生长也表现出显著的治疗效果。这些结果表明,AZD3463是治疗NB的一种有前景的治疗药物。