Tari Crochet Gamze, Ari-Yuka Selcen, Fischer Anja, Chour Mohamed, Claudel Alexis, Sako Nouhoum, Robe Cyrielle, Naudet Julie, Gonon Alexis, Mboumba Diana Laure, Ortonne Nicolas, Alcazer Vincent, Delfau-Larue Marie-Hélène, Siebert Reiner, Gaulard Philippe, Lemonnier François
Paris Est-Créteil University, Créteil, France.
Mondor Biomedical Research Institute, Team Ortonne, INSERM U955, Créteil, France.
Leukemia. 2025 May 20. doi: 10.1038/s41375-025-02628-0.
Follicular helper T-cell lymphoma (TFHL) is the most common non-cutaneous T-cell lymphoma in the Western world and is associated with a poor prognosis. Neoplastic cells rely heavily on the tumor microenvironment, demonstrated by the absence of TFHL-derived cell lines, which hinders therapeutic progress. To overcome this limitation, we developed and characterized patient-derived xenograft TFHL (TFHL-PDXs). Fifteen TFHLs were implanted into immunodeficient mice, generating nine PDXs. The tumor microenvironment was detected in the first passage but progressively disappeared in subsequent passages. TET2 mutations persisted in all cases and TFHL-specific mutations were observed in most. The models were treated with azacitidine and patient sensitivity was fully recapitulated. To elucidate the mechanism of action of azacitidine, we analyzed the differences in DNA methylation and gene expression in six TFHL-PDX models. Global DNA hypomethylation occurred in azacitidine-treated cells in drug-sensitive models but not in the resistant ones. DNA hypomethylation was associated with global upregulation of gene expression, including that of various cancer-related pathways, suggestive of p53-pathway-mediated cytotoxicity. Overall, the PDXs recapitulated TFHL features and exhibited sensitivity to azacitidine. They also made it possible to decipher the mechanism responsible for the effect of azacitidine, revealing the activation of p53-mediated apoptosis associated with DNA hypomethylation.
滤泡辅助性T细胞淋巴瘤(TFHL)是西方世界最常见的非皮肤性T细胞淋巴瘤,且预后较差。肿瘤细胞严重依赖肿瘤微环境,这一点从缺乏TFHL衍生的细胞系便可看出,而这也阻碍了治疗进展。为克服这一限制,我们构建并鉴定了患者来源的异种移植TFHL(TFHL-PDXs)。将15例TFHL植入免疫缺陷小鼠体内,成功构建了9个PDX模型。在传代过程中首次检测到肿瘤微环境,但在后续传代中逐渐消失。所有病例中TET2突变均持续存在,且大多数病例中观察到了TFHL特异性突变。用阿扎胞苷对这些模型进行治疗,完全重现了患者的敏感性。为阐明阿扎胞苷的作用机制,我们分析了6个TFHL-PDX模型中DNA甲基化和基因表达的差异。在药物敏感模型中,阿扎胞苷处理的细胞发生了全基因组DNA低甲基化,而耐药模型中则未出现。DNA低甲基化与基因表达的整体上调相关,包括各种癌症相关通路的基因表达上调,提示p53通路介导的细胞毒性作用。总体而言,PDX模型重现了TFHL的特征,并对阿扎胞苷表现出敏感性。它们还使得解读阿扎胞苷作用效果的机制成为可能,揭示了与DNA低甲基化相关的p53介导的细胞凋亡的激活。