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先天性噬血细胞性淋巴组织细胞增生症(HLH)。

Angeborene hämophagozytische Lymphohistiozytose (HLH).

作者信息

Pachlopnik Schmid J, de Saint Basile G

机构信息

Children's Hospital, University of Zurich, Steinwiesstrasse 75, Zurich, Switzerland.

出版信息

Klin Padiatr. 2010 Nov;222(6):345-50. doi: 10.1055/s-0029-1246165. Epub 2010 May 10.

Abstract

Hemophagocytic lymphohistiocytosis (HLH) is a potentially fatal immune disorder characterized by uncontrolled lymphocyte- and macrophage-activation. The resulting hypercytokinemia and cell infiltration of organs lead to the clinical and laboratory features of HLH. Viral infections and other triggers can induce both, inherited and acquired forms of HLH. Disease-causing mutations in the genes encoding perforin (PRF1, FHL2), munc13-4 (UNC13D, FHL3), syntaxin 11 (STX11, FHL4), and munc18-2 (UNC18-2/STXBP2, FHL5) have been previously identified in Familial Hemophagocyic Lymphohistiocytosis (FHL), whereas mutation in RAB27A and LYST account for Griscelli syndome type 2 and Chediak-Higashi syndrome, respectively. These genes all encode proteins which are involved in the cytotoxic activity of lymphocytes. The inability of activated cytotoxic cells to clear antigen-presenting targets results in sustained immune stimulation, likely accounting for the unremitting polyclonal CD8 T-cell activation and hyperimmune reaction which characterizes FHL. Treatment of HLH consists of elimination of the trigger and immunosuppressive treatment in order to induce remission from the uncontrolled inflammation. Allogeneic hematopoietic stem cell transplantation can be indicated in the inherited forms of HLH.

摘要

噬血细胞性淋巴组织细胞增生症(HLH)是一种潜在致命的免疫紊乱疾病,其特征为淋巴细胞和巨噬细胞的激活失控。由此产生的高细胞因子血症和器官的细胞浸润导致了HLH的临床和实验室特征。病毒感染和其他触发因素可诱发遗传性和获得性HLH。先前已在家族性噬血细胞性淋巴组织细胞增生症(FHL)中鉴定出编码穿孔素(PRF1,FHL2)、munc13 - 4(UNC13D,FHL3)、 syntaxin 11(STX11,FHL4)和munc18 - 2(UNC18 - 2/STXBP2,FHL5)的基因中的致病突变,而RAB27A和LYST的突变分别导致2型格里塞利综合征和切迪阿克 - 东综合征。这些基因均编码参与淋巴细胞细胞毒性活性的蛋白质。活化的细胞毒性细胞无法清除抗原呈递靶标导致持续的免疫刺激,这可能是FHL特征性的持续性多克隆CD8 T细胞活化和高免疫反应的原因。HLH的治疗包括消除触发因素和进行免疫抑制治疗,以诱导从失控的炎症中缓解。异基因造血干细胞移植可用于遗传性HLH的治疗。

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