Nguyen Trang Tt, Torrini Consuelo, Shang Enyuan, Shu Chang, Mun Jeong-Yeon, Gao Qiuqiang, Humala Nelson, Akman Hasan O, Zhang Guoan, Westhoff Mike-Andrew, Karpel-Massler Georg, Bruce Jeffrey N, Canoll Peter, Siegelin Markus D
Department of Pathology and Cell Biology, Columbia University Medical Center, New York, New York, USA.
Department of Biological Sciences, Bronx Community College, City University of New York, New York, USA.
JCI Insight. 2024 Mar 14;9(8):e172565. doi: 10.1172/jci.insight.172565.
Glioblastoma (GBM) remains an incurable disease, requiring more effective therapies. Through interrogation of publicly available CRISPR and RNAi library screens, we identified the α-ketoglutarate dehydrogenase (OGDH) gene, which encodes an enzyme that is part of the tricarboxylic acid (TCA) cycle, as essential for GBM growth. Moreover, by combining transcriptome and metabolite screening analyses, we discovered that loss of function of OGDH by the clinically validated drug compound CPI-613 was synthetically lethal with Bcl-xL inhibition (genetically and through the clinically validated BH3 mimetic, ABT263) in patient-derived xenografts as well neurosphere GBM cultures. CPI-613-mediated energy deprivation drove an integrated stress response with an upregulation of the BH3-only domain protein, Noxa, in an ATF4-dependent manner, as demonstrated by genetic loss-of-function experiments. Consistently, silencing of Noxa attenuated cell death induced by CPI-613 in model systems of GBM. In patient-derived xenograft models of GBM in mice, the combination treatment of ABT263 and CPI-613 suppressed tumor growth and extended animal survival more potently than each compound on its own. Therefore, combined inhibition of Bcl-xL along with disruption of the TCA cycle might be a treatment strategy for GBM.
胶质母细胞瘤(GBM)仍然是一种无法治愈的疾病,需要更有效的治疗方法。通过对公开可用的CRISPR和RNAi文库筛选进行研究,我们确定了α-酮戊二酸脱氢酶(OGDH)基因,该基因编码一种作为三羧酸(TCA)循环一部分的酶,对GBM的生长至关重要。此外,通过结合转录组和代谢物筛选分析,我们发现临床验证的药物化合物CPI-613导致的OGDH功能丧失,在患者来源的异种移植以及神经球GBM培养物中,与Bcl-xL抑制(通过基因手段以及临床验证的BH3模拟物ABT263)具有合成致死性。如基因功能丧失实验所示,CPI-613介导的能量剥夺以ATF4依赖的方式驱动了一种综合应激反应,导致仅含BH3结构域的蛋白Noxa上调。同样,在GBM模型系统中,沉默Noxa可减弱CPI-613诱导的细胞死亡。在小鼠患者来源的GBM异种移植模型中,ABT263和CPI-613联合治疗比单独使用每种化合物更有效地抑制肿瘤生长并延长动物存活时间。因此,联合抑制Bcl-xL以及破坏TCA循环可能是GBM的一种治疗策略。