Molecular Pathology of Lymphoid Neoplasms, Fundació Clínic per a la Recerca Biomèdica - Institut d'Investigacions Biomèdiques August Pi i Sunyer, Barcelona, Spain.
Universitat de Barcelona, Spain.
Blood Adv. 2023 Jul 25;7(14):3695-3709. doi: 10.1182/bloodadvances.2022009415.
The genetic mechanisms associated with splenic marginal zone lymphoma (SMZL) transformation are not well defined. We studied 41 patients with SMZL that eventually underwent large B-cell lymphoma transformation. Tumor material was obtained either only at diagnosis (9 patients), at diagnosis and transformation (18 patients), and only at transformation (14 patients). Samples were categorized in 2 groups: (1) at diagnosis (SMZL, n = 27 samples), and (2) at transformation (SMZL-T, n = 32 samples). Using copy number arrays and a next-generation sequencing custom panel, we identified that the main genomic alterations in SMZL-T involved TNFAIP3, KMT2D, TP53, ARID1A, KLF2, 1q gains, and losses of 9p21.3 (CDKN2A/B) and 7q31-q32. Compared with SMZL, SMZL-T had higher genomic complexity, and higher incidence of TNFAIP3 and TP53 alterations, 9p21.3 (CDKN2A/B) losses, and 6p gains. SMZL and SMZL-T clones arose by divergent evolution from a common altered precursor cell that acquired different genetic alterations in virtually all evaluable cases (92%, 12 of 13 cases). Using whole-genome sequencing of diagnostic and transformation samples in 1 patient, we observed that the SMZL-T sample carried more genomic aberrations than the diagnostic sample, identified a translocation t(14;19)(q32;q13) present in both samples, and detected a focal B2M deletion due to chromothripsis acquired at transformation. Survival analysis showed that KLF2 mutations, complex karyotype, and International Prognostic Index score at transformation were predictive of a shorter survival from transformation (P = .001; P = .042; and P = .007; respectively). In summary, SMZL-T are characterized by higher genomic complexity than SMZL, and characteristic genomic alterations that could represent key players in the transformation event.
与脾边缘区淋巴瘤 (SMZL) 转化相关的遗传机制尚不清楚。我们研究了 41 例最终发生大 B 细胞淋巴瘤转化的 SMZL 患者。肿瘤标本分别仅在诊断时(9 例)、诊断时和转化时(18 例)以及仅在转化时(14 例)获得。样本分为 2 组:(1)诊断时(SMZL,n=27 个样本),(2)转化时(SMZL-T,n=32 个样本)。使用拷贝数阵列和下一代测序定制面板,我们发现 SMZL-T 的主要基因组改变涉及 TNFAIP3、KMT2D、TP53、ARID1A、KLF2、1q 增益和 9p21.3(CDKN2A/B)和 7q31-q32 的缺失。与 SMZL 相比,SMZL-T 的基因组复杂性更高,TNFAIP3 和 TP53 改变、9p21.3(CDKN2A/B)缺失和 6p 增益的发生率更高。SMZL 和 SMZL-T 克隆是由共同的改变前体细胞通过不同的进化途径产生的,几乎所有可评估的病例(92%,13 例中的 12 例)都获得了不同的遗传改变。对 1 例患者的诊断和转化样本进行全基因组测序后,我们观察到 SMZL-T 样本比诊断样本携带更多的基因组异常,在两个样本中均检测到存在 t(14;19)(q32;q13)易位,并检测到由于转化时获得的染色体碎裂而导致的焦点 B2M 缺失。生存分析显示,KLF2 突变、复杂核型和转化时的国际预后指数评分是预测从转化到死亡的生存时间更短的因素(P=0.001;P=0.042;P=0.007;分别)。总之,SMZL-T 的特点是比 SMZL 具有更高的基因组复杂性,以及特征性的基因组改变,这些改变可能代表转化事件中的关键因素。