Pray Betsy, Baiocchi Ethan, Leon Sydney, Barta Brian, Koirala Shirsha, Tsyba Liudmyla, Hinterschied Claire, Carvajal-Moreno Jessika, Hout Ian, Nishat Shamama, Jindal Udita, Jain Neeraj, Singh Satishkumar, Sehgal Lalit, Chan Wing Keung, Hanel Walter, Yalowich Jack, Baiocchi Robert A, Alinari Lapo
Department of Veterinary Biosciences, College of Veterinary Medicine, The Ohio State University, Columbus, OH.
Division of Hematology, Department of Internal Medicine, James Comprehensive Cancer Center, The Ohio State University, Columbus, OH.
Blood Adv. 2025 Apr 22;9(8):2006-2018. doi: 10.1182/bloodadvances.2024015769.
Mantle cell lymphoma (MCL) is an incurable B-cell lymphoma characterized by significant genomic instability. Patients with MCL who progress on targeted therapies have a short survival; thus, novel therapeutic strategies are urgently needed. Overexpression of transducin β-like protein 1 X-linked (TBL1X) has been documented in several types of cancer and associated with poor prognosis. TBL1X is a critical regulator of multiple oncogenic networks; however, its function in MCL has not been explored. Our data show that, unlike normal B cells, MCL cells express abundant levels of TBL1X and that genetic knockdown of TBL1X and treatment with tegavivint (Iterion), a first-in-class small molecule targeting TBL1X, promote MCL cell death in vitro and in vivo. Moreover, TBL1X controls the stability of key MCL oncogenic drivers, cyclin D1 and RAD51; and targeting TBL1X results in significant DNA damage, cell cycle arrest, and ultimately cell death. Combining tegavivint with poly(adenosine 5'-diphosphate-ribose) polymerase-1/2 inhibitor talazoparib results in synergistic MCL cell death in vitro, and in vivo this combination significantly prolongs the survival of a patient-derived MCL xenograft. Together, our results define the role of TBL1X in maintaining genomic stability in MCL and establish targeting TBL1X as a novel therapeutic strategy for patients with this incurable disease.
套细胞淋巴瘤(MCL)是一种无法治愈的B细胞淋巴瘤,其特征是具有显著的基因组不稳定性。接受靶向治疗后病情进展的MCL患者生存期较短;因此,迫切需要新的治疗策略。转导素β样蛋白1 X连锁(TBL1X)在多种癌症类型中均有过表达,且与预后不良相关。TBL1X是多个致癌网络的关键调节因子;然而,其在MCL中的功能尚未得到探索。我们的数据表明,与正常B细胞不同,MCL细胞表达大量的TBL1X,并且TBL1X的基因敲低以及使用首个靶向TBL1X的小分子药物替加维文(Iterion)进行治疗,均可在体外和体内促进MCL细胞死亡。此外,TBL1X控制着MCL关键致癌驱动因子细胞周期蛋白D1和RAD51的稳定性;靶向TBL1X会导致显著的DNA损伤、细胞周期停滞,并最终导致细胞死亡。将替加维文与聚(腺苷5'-二磷酸-核糖)聚合酶-1/2抑制剂他拉唑帕尼联合使用,可在体外协同诱导MCL细胞死亡,在体内这种联合用药可显著延长患者来源的MCL异种移植瘤的生存期。总之,我们的研究结果明确了TBL1X在维持MCL基因组稳定性中的作用,并确立了靶向TBL1X作为这种无法治愈疾病患者的一种新治疗策略。