Kim AeRang, Pratilas Christine A
Children's National Medical Center, Washington, D.C., United States.
Sidney Kimmel Comprehensive Cancer Center at Johns Hopkins, Baltimore, MD, United States.
Exp Neurol. 2018 Jan;299(Pt B):317-325. doi: 10.1016/j.expneurol.2017.08.014. Epub 2017 Aug 30.
Neurofibromatosis type 1 (NF1) is an autosomal dominant tumor predisposition syndrome. Malignant peripheral nerve sheath tumors (MPNST) are aggressive soft tissue sarcomas arising from peripheral nerve sheaths, and the most commonly lethal feature associated with NF1. The hallmark of NF1 and NF1-related MPNST is the loss of neurofibromin expression. Loss of neurofibromin is considered a tumor-promoting event, and leads to constitutive activation of RAS and its downstream effectors. However, RAS activation alone is not sufficient for MPNST formation, and additional tumor suppressors and signaling pathways have been implicated in tumorigenesis of MPNST. Taking advantage of the rapid development of novel therapeutics targeting key molecular pathways across all cancer types, the best-in-class modulators of these pathways can be assessed in pre-clinical models and translated into clinical trials for patients with MPNST. Here, we describe the genetic changes and molecular pathways that drive MPNST formation and highlight the promise of signal transduction to identify therapies that may treat these tumors more effectively.
1型神经纤维瘤病(NF1)是一种常染色体显性肿瘤易感综合征。恶性外周神经鞘瘤(MPNST)是起源于外周神经鞘的侵袭性软组织肉瘤,也是与NF1相关的最常见致死特征。NF1及与NF1相关的MPNST的标志是神经纤维瘤蛋白表达缺失。神经纤维瘤蛋白缺失被认为是一种促肿瘤事件,会导致RAS及其下游效应器的组成性激活。然而,仅RAS激活不足以形成MPNST,其他肿瘤抑制因子和信号通路也与MPNST的肿瘤发生有关。利用针对所有癌症类型关键分子通路的新型疗法的快速发展,可以在临床前模型中评估这些通路的最佳调节剂,并转化为针对MPNST患者的临床试验。在此,我们描述了驱动MPNST形成的基因变化和分子通路,并强调了信号转导在识别可能更有效治疗这些肿瘤的疗法方面的前景。