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首页> 外文期刊>Journal of Neuroimmunology: Official Bulletin of the Research Committee on Neuroimmunology of the World Federation of Neurology >Evidence of recurrent atypical meningioma with rhabdoid transformation and expression of pyrogenic cytokines in a child presenting with a marked acute-phase response: case report and review of the literature.
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Evidence of recurrent atypical meningioma with rhabdoid transformation and expression of pyrogenic cytokines in a child presenting with a marked acute-phase response: case report and review of the literature.

机译：表现为急性期反应的儿童复发性非典型脑膜瘤伴横纹肌转化和热原细胞因子表达的证据：病例报告和文献复习。

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Children presenting with acute systemic illnesses that lack specific clinical or serological defining features may be diagnosed as having a chronic infection, an atypical systemic vasculitis or a connective tissue disease, but often turn out to have occult neoplasias. Cytokines have been implicated in causing many of the systemic effects in such cases. In this study, we describe the case of a 9-year-old boy presenting at an interval of 18 months with a marked acute-phase response due to a recurrent atypical meningioma with rhabdoid transformation of the tentorium cerebelli. Resection of the recurrent tumor was curative. We evaluated in detail the local and systemic production of cytokines released by the primary and the recurrent tumor. Blood and CSF samples were taken pre-, intra-, and postoperatively, and the production of IL-6, IL-1beta, and TNF-alpha was measured by enzyme-linked immunosorbent assays (ELISA). The level of IL-6 in CSF was about 150-fold increased before tumor resection, normalizing postoperatively. On the contrary, the levels of IL-1beta and TNF-alpha in CSF and of IL-6, IL-1beta, and TNF-alpha in serum were pre-, intra-, and postoperatively within normal limits. Cytokine production was also evaluated immunohistochemically, and confirmed strong IL-6 and TNF-alpha expression in the primary and the recurrent tumor, while expression of IL-1beta was lacking. The scattered MHC class II- and leukocyte common antigen (LCA)-expressing inflammatory cells, which were infiltrating exclusively the tumoral stroma, had no detectable cytokine immunoreactivity. We conclude that chronic IL-6 and TNF-alpha production by the tumor cells in this patient was responsible for the severe systemic illness with which he presented.
机译：表现出缺乏特定临床或血清学明确特征的急性全身性疾病的儿童可被诊断为患有慢性感染，非典型性全身性血管炎或结缔组织病，但往往被发现患有隐匿性肿瘤。在这种情况下，细胞因子与引起许多全身作用有关。在这项研究中，我们描述了一个9岁男孩的病例，该男孩间隔18个月因复发性非典型脑膜瘤伴小脑腱的横纹肌转化而出现明显的急性期反应。复发性肿瘤切除术治愈。我们详细评估了原发性和复发性肿瘤释放的细胞因子的局部和全身产生。术前，术中和术后均采集血液和脑脊液样品，并通过酶联免疫吸附试验（ELISA）测量IL-6，IL-1beta和TNF-α的产生。肿瘤切除之前，脑脊液中的IL-6水平增加了约150倍，术后恢复正常。相反，脑脊液中IL-1beta和TNF-α的水平以及血清中IL-6，IL-1beta和TNF-α的水平在术前，术中和术后均在正常范围内。还通过免疫组织化学评估了细胞因子的产生，并证实了原发性和复发性肿瘤中强烈的IL-6和TNF-α表达，而缺乏IL-1β表达。散布的仅表达肿瘤基质的表达MHC II类和白细胞共同抗原（LCA）的炎症细胞没有可检测到的细胞因子免疫反应性。我们得出的结论是，该患者中肿瘤细胞产生的慢性IL-6和TNF-α导致了他所表现出的严重全身性疾病。

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