Loehr Amanda R, Timmerman Dennis M, Liu Michelle, Gillis Ad J M, Matthews Melia, Bloom Jordana C, Nicholls Peter K, Page David C, Miller Andrew D, Looijenga Leendert H J, Weiss Robert S
Department of Biomedical Sciences, Cornell University College of Veterinary Medicine, Ithaca, NY.
Princess Máxima Center for Pediatric Oncology, Utrecht, Netherlands.
bioRxiv. 2023 Sep 12:2023.09.09.556995. doi: 10.1101/2023.09.09.556995.
Malignant testicular germ cells tumors (TGCTs) are the most common solid cancers in young men. Current TGCT diagnostics include conventional serum protein markers, but these lack the sensitivity and specificity to serve as accurate markers across all TGCT subtypes. MicroRNAs (miRNAs) are small non-coding regulatory RNAs and informative biomarkers for several diseases. In humans, miRNAs of the miR-371-373 cluster are detectable in the serum of patients with malignant TGCTs and outperform existing serum protein markers for both initial diagnosis and subsequent disease monitoring. We previously developed a genetically engineered mouse model featuring malignant mixed TGCTs consisting of pluripotent embryonal carcinoma (EC) and differentiated teratoma that, like the corresponding human malignancies, originate in utero and are highly chemosensitive. Here, we report that miRNAs in the mouse miR-290-295 cluster, homologs of the human miR-371-373 cluster, were detectable in serum from mice with malignant TGCTs but not from tumor-free control mice or mice with benign teratomas. miR-291-293 were expressed and secreted specifically by pluripotent EC cells, and expression was lost following differentiation induced by the drug thioridazine. Notably, miR-291-293 levels were significantly higher in the serum of pregnant dams carrying tumor-bearing fetuses compared to that of control dams. These findings reveal that expression of the miR-290-295 and miR-371-373 clusters in mice and humans, respectively, is a conserved feature of malignant TGCTs, further validating the mouse model as representative of the human disease. These data also highlight the potential of serum miR-371-373 assays to improve patient outcomes through early TGCT detection, possibly even prenatally.
恶性睾丸生殖细胞肿瘤(TGCTs)是年轻男性中最常见的实体癌。目前TGCT的诊断方法包括传统的血清蛋白标志物,但这些标志物缺乏在所有TGCT亚型中作为准确标志物所需的敏感性和特异性。微小RNA(miRNAs)是小型非编码调节RNA,是多种疾病的信息性生物标志物。在人类中,miR-371-373簇的miRNAs在恶性TGCT患者的血清中可检测到,并且在初始诊断和后续疾病监测方面均优于现有的血清蛋白标志物。我们之前开发了一种基因工程小鼠模型,其特征是具有由多能胚胎癌(EC)和分化型畸胎瘤组成的恶性混合TGCTs,与相应的人类恶性肿瘤一样,起源于子宫内且对化疗高度敏感。在此,我们报告小鼠miR-290-295簇中的miRNAs(人类miR-371-373簇的同源物)在患有恶性TGCTs的小鼠血清中可检测到,但在无肿瘤对照小鼠或患有良性畸胎瘤的小鼠血清中未检测到。miR-291-293由多能EC细胞特异性表达和分泌,并且在硫利达嗪诱导分化后表达消失。值得注意的是,与对照母鼠相比,携带患肿瘤胎儿的怀孕母鼠血清中的miR-291-293水平显著更高。这些发现表明,miR-290-295和miR-371-373簇分别在小鼠和人类中的表达是恶性TGCTs的一个保守特征,进一步验证了该小鼠模型可作为人类疾病的代表。这些数据还突出了血清miR-371-373检测通过早期TGCT检测(甚至可能在产前)改善患者预后的潜力。