Department of Ophthalmology, Shanghai Key Laboratory of Orbital Diseases and Ocular Oncology, Ninth People's Hospital, Shanghai JiaoTong University School of Medicine, Shanghai, P.R. China.
Invest Ophthalmol Vis Sci. 2024 Jun 3;65(6):8. doi: 10.1167/iovs.65.6.8.
Optic pathway gliomas (OPGs) are most predominant pilocytic astrocytomas, which are typically diagnosed within the first decade of life. The majority of affected children with OPGs also present with neurofibromatosis type 1 (NF1), the most common tumor predisposition syndrome. OPGs in individuals with NF1 primarily affect the optic pathway and lead to visual disturbance. However, it is challenging to assess risk in asymptomatic patients without valid biomarkers. On the other hand, for symptomatic patients, there is still no effective treatment to prevent or recover vision loss. Therefore, this review summarizes current knowledge regarding the pathogenesis of NF1-associated OPGs (NF1-OPGs) from preclinical studies to seek potential prognostic markers and therapeutic targets. First, the loss of the NF1 gene activates 3 distinct Ras effector pathways, including the PI3K/AKT/mTOR pathway, the MEK/ERK pathway, and the cAMP pathway, which mediate glioma tumorigenesis. Meanwhile, non-neoplastic cells from the tumor microenvironment (microglia, T cells, neurons, etc.) also contribute to gliomagenesis via various soluble factors. Subsequently, we investigated potential genetic risk factors, molecularly targeted therapies, and neuroprotective strategies for tumor prevention and vision recovery. Last, potential directions and promising preclinical models of NF1-OPGs are presented for further research. On the whole, NF1-OPGs develop as a result of the interaction between glioma cells and the tumor microenvironment. Developing effective treatments require a better understanding of tumor molecular characteristics, as well as multistage interventions targeting both neoplastic cells and non-neoplastic cells.
视神经胶质瘤(OPG)是最常见的毛细胞型星形细胞瘤,通常在生命的第一个十年内被诊断出来。大多数患有 OPG 的受影响儿童也患有神经纤维瘤病 1 型(NF1),这是最常见的肿瘤易感性综合征。NF1 患者的 OPG 主要影响视神经通路,并导致视觉障碍。然而,在没有有效生物标志物的无症状患者中,评估风险具有挑战性。另一方面,对于有症状的患者,仍然没有有效的治疗方法来预防或恢复视力丧失。因此,本综述总结了从临床前研究到寻找潜在预后标志物和治疗靶点的 NF1 相关 OPG(NF1-OPG)发病机制的当前知识。首先,NF1 基因的缺失会激活 3 种不同的 Ras 效应途径,包括 PI3K/AKT/mTOR 途径、MEK/ERK 途径和 cAMP 途径,这些途径介导胶质瘤的发生。同时,肿瘤微环境中的非肿瘤细胞(小胶质细胞、T 细胞、神经元等)也通过各种可溶性因子促进胶质瘤的发生。随后,我们研究了潜在的遗传风险因素、针对分子靶点的治疗方法和神经保护策略,以预防肿瘤和恢复视力。最后,提出了 NF1-OPG 的潜在研究方向和有前景的临床前模型。总的来说,NF1-OPG 的发生是胶质瘤细胞与肿瘤微环境相互作用的结果。开发有效的治疗方法需要更好地了解肿瘤的分子特征,以及针对肿瘤细胞和非肿瘤细胞的多阶段干预。