Fatal Coxsackie meningoencephalitis in a patient with B-cell lymphopenia and hypogammaglobulinemia following rituximab therapy.

Fatal Coxsackie meningoencephalitis in a patient with B-cell lymphopenia and hypogammaglobulinemia following rituximab therapy.
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利妥昔单抗治疗后出现 B 细胞淋巴细胞减少和低丙种球蛋白血症的患者发生致命柯萨奇脑膜脑炎。

DOI:
10.1016/j.anai.2015.05.007
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发表时间:
2015
期刊:
Annals of allergy, asthma & immunology : official publication of the American College of Allergy, Asthma, & Immunology
影响因子:
--
通讯作者:
Lawrence,Monica
Lawrence,Monica
中科院分区:
--
文献类型:
--
作者:
Palacios,Thamiris;Bartelt,Luther;Scheld,William;Lopes,MBeatriz;Kelting,SarahM;Holland,Steven;Lipkin,WIan;Quan,Phenix-Lan;Borish,Larry;Lawrence,Monica

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肠道病毒(EV)是最常见的循环病毒,也是美国病毒性脑膜脑炎的病因。在一般人群中,肠道病毒感染通常是一种自限性疾病。然而,在体液免疫缺陷患者中,感染可能是致命的。本病例是一名患有埃文斯综合征的妇女,经脾切除术和利妥昔单抗治疗后出现低丙种球蛋白血症,尽管进行了抗体替代,但仍发生了致命的科萨基B3病毒脑膜脑炎。虽然EV脑膜脑炎(EM)通常是自限性的,但在体液免疫缺陷患者中,慢性EM经常报告致命结局。静脉注射免疫球蛋白(IVIG)可以显著降低先天性无丙种球蛋白血症患者慢性EM的发生率。不幸的是,一旦慢性EM发展,可用的药物治疗选择有限,所有治疗都有不同程度的成功,包括普来考那利(一种小核糖核酸病毒衣壳蛋白抑制剂),高剂量IVIG,鞘内免疫球蛋白,以及使用选择含有高滴度肠道病毒抗体的免疫球蛋白制剂。
Enteroviruses (EVs) are the most common circulating viruses and cause of viral meningoencephalitis in the United States. In the general population, enteroviral infection is usually a self-limited disease. However, in patients with humoral immunodeficiency, infections can be fatal. The present case is that of a woman with Evans syndrome treated with splenectomy and rituximab who developed hypogammaglobulinemia and, despite antibody replacement, a fatal Coxsackie B3 virus meningoencephalitis.Enteroviruses are positive-sense single-stranded RNA viruses with a capsid composed of 4 structural proteins (VP1eVP4). 1, 2 Although EV meningoencephalitis (EM) is typically selflimited, in patients with humoral immunodeficiency, chronic EM has been reported often with fatal outcome. 3, 4 The use of intravenous immunoglobulin (IVIG) has dramatically decreased the incidence of chronic EM in congenital agammaglobulinemic conditions. Unfortunately, there are limited pharmaceutical treatment options available once chronic EM develops, all with varying degrees of success, including pleconaril (a picornavirus capsid protein inhibitor), high-dose IVIG, intrathecal immunoglobulin, and use of immunoglobulin preparations selected to contain high titers of enteroviral antibody.
无丙种球蛋白血症患者的慢性肠道病毒性脑膜脑炎
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发表时间: 1988
期刊:
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