Zhang Qian, Wang Hong Y, Liu Xiaobin, Roth Michael H, Shestov Alex A, Lee Seung-Cheol, Jain Kanika, Soderquist Craig, Xiong Qun-Bin, Ruella Marco, Strauser Honore, Glickson Jerry D, Schuster Stephen J, Ptasznik Andrzej, Wasik Mariusz A
Department of Pathology and Laboratory Medicine, Abramson Cancer Center, Perelman School of Medicine of the University of Pennsylvania, Philadelphia, PA, United States.
Department of Radiology, Abramson Cancer Center, Perelman School of Medicine of the University of Pennsylvania, Philadelphia, PA, United States.
Front Oncol. 2019 Jul 3;9:568. doi: 10.3389/fonc.2019.00568. eCollection 2019.
While studies have identified a number of mutations in mantle cell lymphoma (MCL), the list may still be incomplete and contribution to the pathogenesis remains unclear. We analyzed the mutational landscape of four mantle cell lymphoma biopsies obtained during an 8-year period from the same patient with his normal cells serving as control; we also established a cell line from the final stage of the disease. Numerous mutations with high allelic burden have been identified in all four biopsies. While a large subset of mutations was seen only in individual biopsies, the core of 21 mutations persisted throughout the disease. This mutational core is also maintained in the cell line that also displays DNA-methylation and cytokine secretion profiles of the primary mantle cell lymphoma cells. This cell line is uniquely sensitive to clinically relevant inhibitors of Bruton's Tyrosine Kinase. The response to Bruton Tyrosine Kinase's inhibition is enhanced by inhibitors of CDK4/6 and mTOR. Among the mutations seen in the primary and cultured MCL cells, mutations of three genes are involved in the control of H3K4 methylation: demethylase KDM5C, present already in the early disease, and methyltransferase KMT2D and cofactor BCOR, both of which are seen late in the disease and are novel and predicted to be pathogenic. The presence of these mutations was associated with hypermethylation of H3K4. Restoration of KDM5C expression affected expression of numerous genes involved in cell proliferation, adherence/movement, and invasiveness.
虽然已有研究确定了套细胞淋巴瘤(MCL)中的一些突变,但该列表可能仍不完整,其对发病机制的贡献仍不清楚。我们分析了一名患者在8年期间获得的4份套细胞淋巴瘤活检样本的突变图谱,以其正常细胞作为对照;我们还从疾病晚期建立了一个细胞系。在所有4份活检样本中均发现了大量具有高等位基因负担的突变。虽然很大一部分突变仅在个别活检样本中出现,但21个核心突变在整个疾病过程中持续存在。这个突变核心在细胞系中也得以保留,该细胞系还显示出原发性套细胞淋巴瘤细胞的DNA甲基化和细胞因子分泌谱。该细胞系对临床上相关的布鲁顿酪氨酸激酶抑制剂具有独特的敏感性。CDK4/6和mTOR抑制剂可增强对布鲁顿酪氨酸激酶抑制的反应。在原发性和培养的MCL细胞中发现的突变中,有三个基因的突变参与了H3K4甲基化的调控:去甲基化酶KDM5C在疾病早期就已存在,甲基转移酶KMT2D和辅因子BCOR在疾病晚期出现,二者均为新发现且预计具有致病性。这些突变的存在与H3K4的高甲基化有关。恢复KDM5C的表达会影响许多参与细胞增殖、黏附/运动和侵袭的基因的表达。