Université d'Angers, Inserm, CRCINA, Angers, France.
Unité de Formation et de Recherche (UFR) Santé, Université d'Angers, Angers, France.
Blood Adv. 2020 Oct 13;4(19):4887-4897. doi: 10.1182/bloodadvances.2020002271.
Among myeloproliferative neoplasms, polycythemia vera (PV) and essential thrombocythemia (ET) are the 2 entities associated with the most chronic disease course. Leukemic evolution occurs rarely but has a grim prognosis. The interval between diagnosis and leukemic evolution is highly variable, from a few years to >20 years. We performed a molecular evaluation of 49 leukemic transformations of PV and ET by targeted next-generation sequencing. Using a hierarchical classification, we identified 3 molecular groups associated with a distinct time to leukemic transformation. Short-term transformations were mostly characterized by a complex molecular landscape and mutations in IDH1/2, RUNX1, and U2AF1 genes, whereas long-term transformations were associated with mutations in TP53, NRAS, and BCORL1 genes. Studying paired samples from chronic phase and transformation, we detected some mutations already present during the chronic phase, either with a significant allele burden (short-term transformation) or with a very low allele burden (especially TP53 mutations). However, other mutations were not detected even 1 year before leukemic transformation. Our results suggest that the leukemic transformation of PV and ET may be driven by distinct time-dependent molecular mechanisms.
在骨髓增殖性肿瘤中,真性红细胞增多症(PV)和原发性血小板增多症(ET)是与病程最慢性相关的两种疾病。白血病转化虽然罕见,但预后较差。从诊断到白血病转化的间隔时间差异很大,从几年到超过 20 年不等。我们通过靶向下一代测序对 49 例 PV 和 ET 的白血病转化进行了分子评估。通过分层分类,我们确定了 3 个与白血病转化时间明显相关的分子群。短期转化主要表现为复杂的分子谱和 IDH1/2、RUNX1 和 U2AF1 基因突变,而长期转化与 TP53、NRAS 和 BCORL1 基因突变相关。研究慢性期和转化期的配对样本,我们发现了一些在慢性期就已经存在的突变,要么具有显著的等位基因负担(短期转化),要么具有非常低的等位基因负担(尤其是 TP53 突变)。然而,即使在白血病转化前 1 年,也未检测到其他突变。我们的结果表明,PV 和 ET 的白血病转化可能由不同的、随时间变化的分子机制驱动。