Sjogren's syndrome-associated meningoencephalomyelitis: Cerebrospinal fluid cytokine levels and therapeutic utility of tacrolimus

Sjogren's syndrome-associated meningoencephalomyelitis: Cerebrospinal fluid cytokine levels and therapeutic utility of tacrolimus
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DOI:
10.1016/j.jns.2007.10.009
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发表时间:
2008-04-15
影响因子:
4.4
通讯作者:
Hara, Toshiro
Hara, Toshiro
中科院分区:
医学3区
文献类型:
--
作者:
Hoshina, Takayuki;Yamaguchi, Yui;Hara, Toshiro

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对一例干燥综合征(SS)相关性脑膜脑脊髓炎患者的循环和脑脊液(CSF)细胞因子水平的系列变化进行了评估。一个16岁的女孩被诊断为原发性SS在8岁的头痛和呕吐。脑脊液检查显示淋巴细胞占主导地位的白细胞增多和高IgM指数,但没有感染的证据。意识障碍和脑电图弥漫性慢波导致诊断为S -脑膜脑炎。经过一个周期的地塞米松治疗后,临床症状缓解,但2个月后出现尿潴留和下半身感觉异常。颅脊髓磁共振成像(MRI)显示广泛的实质内病变,T2加权高信号强度邻近大脑侧脑室左后角和脊髓C5至T10的纵向病变。大剂量甲基强的松龙和随后的他克莫司治疗有效地控制了SS-脑膜脑脊髓炎的活动。在疾病过程中监测全身和CSF细胞因子水平显示,CSF白细胞介素-6,而不是干扰素-γ或肿瘤坏死因子-α水平是疾病活动的敏感指标。独特的细胞因子谱,不同于那些感染性脑膜炎可能是有用的预测中枢神经系统参与自身免疫性疾病。(c)2007年由Elsevier B. V.出版。
Serial changes in the circulating and cerebrospinal fluid (CSF) cytokine levels were assessed in a patient with Sjogren's syndrome (SS)associated meningoencephalomyelitis. A 16-yr-old girl diagnosed as having primary SS at 8 yr of age presented headache and vomiting. CSF studies revealed lymphocyte-dominant pleocytosis and high IgM index, but no evidence of infection. Disturbed consciousness and diffuse slow waves on electroencephalogram led to the diagnosis of S S -meningoencephalitis. The clinical condition subsided after a cycle of dexamethasone therapy, however, 2 months later urinary retention and paresthesia of the lower body developed. Craniospinal magnetic resonance imaging (MRI) showed extensive intraparenchymal lesions with high T2-weighted signal intensity adjacent to the posterior left horn of lateral ventricle of the brain and the longitudinal lesion from C5 to T10 of the spinal cord. High-dose methyl-prednisolone and subsequent tacrolimus therapy has effectively controlled the activity of SS-meningoencephalomyelitis. Monitoring of systemic and CSF cytokine levels during the course of illness revealed that CSF interleukin-6, but not interferon-gamma or tumor necrosis factor-alpha levels were the sensitive indicator of disease activity. The unique cytokine profile, differing from those of infectious meningitis may be useful for predicting the central nervous system involvement in autoimmune disease. (c) 2007 Published by Elsevier B.V.