Booman M, Szuhai K, Rosenwald A, Hartmann E, Kluin-Nelemans Hc, de Jong D, Schuuring E, Kluin Pm
Department of Pathology and Laboratory Medicine, University Medical Center Groningen, University of Groningen, Groningen, The Netherlands.
J Pathol. 2008 Oct;216(2):209-17. doi: 10.1002/path.2399.
Primary diffuse large B-cell lymphomas of different immune-privileged sites (IP-DLBCLs) share many clinical and biological features, such as a relatively poor prognosis, preferential dissemination to other immune-privileged sites, and deletion of the HLA region, which suggests that IP-DLBCL represents a separate entity. To further investigate the nature of IP-DLBCL, we investigated site-specific genomic aberrations in 16 testicular, nine central nervous system (CNS), and 15 nodal DLBCLs using array CGH. We also determined minimal common regions of gain and loss. Using robust algorithms including multiple testing procedures and the ACE-it script, which is specifically designed for this task, the array CGH data were combined with gene expression data to explore pathways deregulated by chromosomal aberrations. Loss of 6p21.32-p25.3, including the HLA genes, was associated with both types of IP-DLBCL, whereas gain of 2p16.1-p25.3 was associated with nodal DLBCL. Gain of 12q15-q21.1 and 12q24.32-q24.33 was associated with CNS DLBCL and gain of 19q13.12-q13.43 with testicular DLBCL. Analysis of candidate genes in site-specific regions and minimal common regions revealed two major groups of genes: one involved in the immune response, including regulation of HLA expression, and the other involved in apoptosis, including the p53 pathway. Many of these genes were also involved in homozygous deletions or high-level gains. The presence of both shared and site-specific aberrations in CNS and testicular DLBCLs underlines the concept of IP-DLBCL but also indicates that IP-DLBCLs of the CNS and testis do not form a single entity. The observed aberrations emphasize the importance of the deregulation of anti-tumour immune response and apoptosis pathways.
不同免疫豁免部位的原发性弥漫性大B细胞淋巴瘤(IP-DLBCL)具有许多临床和生物学特征,如预后相对较差、倾向于扩散至其他免疫豁免部位以及HLA区域缺失,这表明IP-DLBCL代表一个独立的实体。为进一步研究IP-DLBCL的本质,我们使用阵列比较基因组杂交技术(array CGH)研究了16例睾丸、9例中枢神经系统(CNS)和15例淋巴结DLBCL中的位点特异性基因组畸变。我们还确定了增减的最小共同区域。使用包括多重检验程序和专门为此任务设计的ACE-it脚本等稳健算法,将阵列CGH数据与基因表达数据相结合,以探索由染色体畸变失调的通路。包括HLA基因在内的6p21.32-p25.3缺失与两种类型的IP-DLBCL相关,而2p16.1-p25.3增益与淋巴结DLBCL相关。12q15-q21.1和12q24.32-q24.33增益与CNS DLBCL相关,19q13.12-q13.43增益与睾丸DLBCL相关。对位点特异性区域和最小共同区域中的候选基因进行分析,发现了两大类基因:一类参与免疫反应,包括HLA表达的调节,另一类参与细胞凋亡,包括p53通路。这些基因中的许多也参与了纯合缺失或高水平增益。CNS和睾丸DLBCL中存在共同和位点特异性畸变,这既强调了IP-DLBCL的概念,也表明CNS和睾丸的IP-DLBCL并非形成一个单一实体。观察到的畸变强调了抗肿瘤免疫反应和细胞凋亡通路失调的重要性。