Ostrow Kimberly Laskie, Donaldson Katelyn, Blakeley Jaishri, Belzberg Allan, Hoke Ahmet
Department of Neurology, The Johns Hopkins School of Medicine, Baltimore, MD, 21205, United States of America.
Deparment of Oncology, The Johns Hopkins School of Medicine, Baltimore, MD, 21205, United States of America.
PLoS One. 2015 Dec 14;10(12):e0144620. doi: 10.1371/journal.pone.0144620. eCollection 2015.
Schwannomatosis, a rare form of neurofibromatosis, is characterized predominantly by multiple, often painful, schwannomas throughout the peripheral nervous system. The current standard of care for schwannomatosis is surgical resection. A major obstacle to schwannomatosis research is the lack of robust tumor cell lines. There is a great need for mechanistic and drug discovery studies of schwannomatosis, yet appropriate tools are not currently available. Schwannomatosis tumors are difficult to grow in culture as they survive only a few passages before senescence. Our lab has extensive experience in establishing primary and immortalized human Schwann cell cultures from normal tissue that retain their phenotypes after immortalization. Therefore we took on the challenge of creating immortalized human Schwann cell lines derived from tumors from schwannomatosis patients. We have established and fully characterized 2 schwannomatosis cell lines from 2 separate patients using SV40 virus large T antigen. One patient reported pain and the other did not. The schwannomatosis cell lines were stained with S100B antibodies to confirm Schwann cell identity. The schwannomatosis cells also expressed the Schwann cell markers, p75NTR, S100B, and NGF after multiple passages. Cell morphology was retained following multiple passaging and freeze/ thaw cycles. Gene expression microarray analysis was used to compare the cell lines with their respective parent tumors. No differences in key genes were detected, with the exception that several cell cycle regulators were upregulated in the schwannomatosis cell lines when compared to their parent tumors. This upregulation was apparently a product of cell culturing, as the schwannomatosis cells exhibited the same expression pattern of cell cycle regulatory genes as normal primary human Schwann cells. Cell growth was also similar between normal primary and immortalized tumor cells in culture. Accurate cell lines derived directly from human tumors will serve as invaluable tools for advancing schwannomatosis research, including drug screening.
神经鞘瘤病是一种罕见的神经纤维瘤病,主要特征是在整个周围神经系统中出现多个常常引起疼痛的神经鞘瘤。目前神经鞘瘤病的标准治疗方法是手术切除。神经鞘瘤病研究的一个主要障碍是缺乏可靠的肿瘤细胞系。对神经鞘瘤病进行机制和药物发现研究的需求很大,但目前尚无合适的工具。神经鞘瘤病肿瘤在培养中很难生长,因为它们在衰老前仅能传代几次就会死亡。我们实验室在从正常组织建立原代和永生化人雪旺细胞培养物方面拥有丰富经验,这些细胞在永生化后仍保留其表型。因此,我们接受了从神经鞘瘤病患者的肿瘤中创建永生化人雪旺细胞系的挑战。我们使用SV40病毒大T抗原从2名不同患者中建立并全面表征了2个神经鞘瘤病细胞系。一名患者报告有疼痛,另一名患者没有。用S100B抗体对神经鞘瘤病细胞系进行染色以确认雪旺细胞身份。经过多次传代后,神经鞘瘤病细胞还表达了雪旺细胞标志物p75NTR、S100B和NGF。经过多次传代以及冻融循环后,细胞形态得以保留。使用基因表达微阵列分析将细胞系与其各自的原发肿瘤进行比较。未检测到关键基因存在差异,不过与它们的原发肿瘤相比,神经鞘瘤病细胞系中有几个细胞周期调节因子上调。这种上调显然是细胞培养的产物,因为神经鞘瘤病细胞表现出与正常原代人雪旺细胞相同的细胞周期调节基因表达模式。培养中的正常原代肿瘤细胞和永生化肿瘤细胞之间的细胞生长情况也相似。直接源自人类肿瘤的准确细胞系将成为推进神经鞘瘤病研究(包括药物筛选)的宝贵工具。