Norvilas Rimvydas, Batiuskaite Raminta, Dirse Vaidas, Semaskeviciene Ruta, Gineikiene Egle, Stoskus Mindaugas, Vaitkeviciene Goda, Rascon Jelena, Griskevicius Laimonas
Hematology, Oncology and Transfusion Medicine Center, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania; Department of Experimental, Preventive and Clinical Medicine, State Research Institute Centre for Innovative Medicine, Vilnius, Lithuania.
Hematology, Oncology and Transfusion Medicine Center, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania.
Leuk Res. 2022 May;116:106825. doi: 10.1016/j.leukres.2022.106825. Epub 2022 Mar 12.
T-cell acute lymphoblastic leukemia (T-ALL) is a genetically diverse disease characterized by a heterogeneous profile of genetic lesions. Recent transcriptome studies identified a number of new T-ALL-related gene fusions. Novel gene fusions could be employed as disease-specific molecular markers and provide a better understanding of T-ALL biology, proving the need for further omics sequencing studies.
We performed a population-based analysis of 65 (26 pediatric and 39 adults) Lithuanian T-ALL patients. Targeted RNA sequencing (RNA-seq) was used to detect recurrent and novel T-ALL-related fusion transcripts and gene sequence variants. RT-qPCR was used to calculate the relative gene expression of fusion transcripts.
We identified disease-related gene alterations in 57/65 (87.7%) T-ALL cases, of which four patients harbored gene fusions that affect ABL-class or JAK-STAT signaling pathways. Five novel gene fusions were detected in 4/65 (6.2%) T-ALL cases: CD99::ABL2 and ZEB1::GNAS in the same case, CTCF::ENKD1, DIAPH1::JAK2, and CDK6::NEK1. Novel fusion transcripts encode chimeric proteins that can potentially affect T-cell proliferation, apoptosis, and help to maintain leukemic cells. Importantly, novel fusion transcripts were not detected in the clinical remission samples attributing these fusions to the leukemic compartment.
We report a similar incidence rate of recurrent gene alterations affecting T-ALL-related molecular signaling pathways in both Lithuanian T-ALL patients and other T-ALL studies. Our data suggest that newly identified gene fusions are specific to leukemic T-cells and provide new molecular insights on T-ALL pathogenesis. Further research is needed to confirm these findings.
T细胞急性淋巴细胞白血病(T-ALL)是一种基因多样的疾病,其特征是基因损伤的异质性特征。最近的转录组研究发现了一些新的T-ALL相关基因融合。新型基因融合可作为疾病特异性分子标志物,有助于更好地理解T-ALL生物学特性,这证明了进一步进行组学测序研究的必要性。
我们对65例(26例儿童和39例成人)立陶宛T-ALL患者进行了基于人群的分析。使用靶向RNA测序(RNA-seq)检测复发性和新型T-ALL相关融合转录本和基因序列变异。RT-qPCR用于计算融合转录本的相对基因表达。
我们在57/65(87.7%)的T-ALL病例中鉴定出与疾病相关的基因改变,其中4例患者存在影响ABL类或JAK-STAT信号通路的基因融合。在4/65(6.2%)的T-ALL病例中检测到5种新型基因融合:同一病例中的CD99::ABL2和ZEB1::GNAS、CTCF::ENKD1、DIAPH1::JAK2以及CDK6::NEK1。新型融合转录本编码嵌合蛋白,这些蛋白可能影响T细胞增殖、凋亡,并有助于维持白血病细胞。重要的是,在临床缓解样本中未检测到新型融合转录本,这表明这些融合与白血病细胞区室有关。
我们报告了立陶宛T-ALL患者和其他T-ALL研究中影响T-ALL相关分子信号通路的复发性基因改变的发生率相似。我们的数据表明,新鉴定的基因融合对白血病T细胞具有特异性,并为T-ALL发病机制提供了新的分子见解。需要进一步研究来证实这些发现。