Sugihara Takahiko, Sekine Chiyoko, Nakae Takashi, Kohyama Kuniko, Harigai Masayoshi, Iwakura Yoichiro, Matsumoto Yoh, Miyasaka Nobuyuki, Kohsaka Hitoshi
Department of Medicine and Rheumatology, Graduate School, Tokyo Medical and Dental University, Tokyo, Japan.
Arthritis Rheum. 2007 Apr;56(4):1304-14. doi: 10.1002/art.22521.
To establish a new murine model of polymyositis (PM) for the understanding of its pathologic mechanisms and the development of new treatment strategies.
C protein-induced myositis (CIM) was induced by a single immunization of recombinant human skeletal C protein in C57BL/6 mice, as well as in CD4-depleted, CD8-depleted, and mutant mice as controls. Some mice were treated with high-dose intravenous immunoglobulin (IVIG) after disease induction. Muscle tissues were examined histologically.
In mice with CIM, inflammation was confined to the skeletal muscles. Histologic examination revealed a common pathologic feature of CIM and PM, involving abundant infiltration of CD8 and perforin-expressing cells in the endomysial site of the injured muscle. Suppression of myositis was achieved by depletion of both CD4 and CD8 T cells. Despite the development of serum anti-C protein antibodies in wild-type mice, severe myositis was induced in mice deficient in B cells. Induction of myositis was suppressed in interleukin-1alpha/beta (IL-1alpha/beta)-null mutant mice, but not in tumor necrosis factor alpha (TNFalpha)-null mutant mice. Use of IVIG, a treatment with proven efficacy in PM, suppressed CIM in the subgroup of treated mice.
CIM mimics PM pathologically and clinically. Infiltration of CD8 T cells is the most likely mechanism of muscle injury, and IL-1, but not B cells or TNFalpha, is crucial in the development of CIM. IVIG has therapeutic effects in CIM, suggesting that the effects of IVIG are not mediated by suppression of antibody-mediated tissue injury. This murine model provides a useful tool for understanding the pathologic mechanisms of PM and for developing new treatment strategies.
建立一种新的多发性肌炎(PM)小鼠模型,以了解其病理机制并开发新的治疗策略。
通过在C57BL/6小鼠中单次免疫重组人骨骼肌C蛋白诱导C蛋白诱导性肌炎(CIM),并以CD4缺失、CD8缺失和突变小鼠作为对照。部分小鼠在疾病诱导后接受大剂量静脉注射免疫球蛋白(IVIG)治疗。对肌肉组织进行组织学检查。
在患有CIM的小鼠中,炎症局限于骨骼肌。组织学检查揭示了CIM和PM的共同病理特征,包括在受损肌肉的肌内膜部位有大量表达CD8和穿孔素的细胞浸润。通过耗尽CD4和CD8 T细胞实现了对肌炎的抑制。尽管野生型小鼠中产生了血清抗C蛋白抗体,但B细胞缺陷的小鼠仍诱导出严重的肌炎。在白细胞介素-1α/β(IL-1α/β)基因敲除突变小鼠中肌炎诱导受到抑制,但在肿瘤坏死因子α(TNFα)基因敲除突变小鼠中未受抑制。使用IVIG(一种在PM中有 proven efficacy的治疗方法)在治疗组小鼠亚组中抑制了CIM。
CIM在病理和临床上模拟PM。CD8 T细胞浸润是最可能的肌肉损伤机制,并且IL-1而非B细胞或TNFα在CIM的发生发展中起关键作用。IVIG在CIM中有治疗作用,表明IVIG的作用不是通过抑制抗体介导的组织损伤来介导的。这种小鼠模型为理解PM的病理机制和开发新的治疗策略提供了一个有用的工具。