Suppr超能文献

Wiskott-Aldrich 综合征:肌动蛋白细胞骨架与免疫细胞功能。

The Wiskott-Aldrich syndrome: The actin cytoskeleton and immune cell function.

机构信息

Molecular Immunology Unit, UCL Institute of Child Health, London, UK.

出版信息

Dis Markers. 2010;29(3-4):157-75. doi: 10.3233/DMA-2010-0735.

Abstract

Wiskott-Aldrich syndrome (WAS) is a rare X-linked recessive primary immunodeficiency characterised by immune dysregulation, microthrombocytopaenia, eczema and lymphoid malignancies. Mutations in the WAS gene can lead to distinct syndrome variations which largely, although not exclusively, depend upon the mutation. Premature termination and deletions abrogate Wiskott-Aldrich syndrome protein (WASp) expression and lead to severe disease (WAS). Missense mutations usually result in reduced protein expression and the phenotypically milder X-linked thrombocytopenia (XLT) or attenuated WAS [1-3]. More recently however novel activating mutations have been described that give rise to X-linked neutropenia (XLN), a third syndrome defined by neutropenia with variable myelodysplasia [4-6]. WASP is key in transducing signals from the cell surface to the actin cytoskeleton, and a lack of WASp results in cytoskeletal defects that compromise multiple aspects of normal cellular activity including proliferation, phagocytosis, immune synapse formation, adhesion and directed migration.

摘要

威特综合征(Wiskott-Aldrich syndrome,WAS)是一种罕见的 X 连锁隐性遗传原发性免疫缺陷病,其特征为免疫失调、微小血小板减少症、湿疹和淋巴样恶性肿瘤。WAS 基因的突变可导致明显的综合征变异,这些变异主要(尽管并非完全)取决于突变。提前终止和缺失使 Wiskott-Aldrich 综合征蛋白(Wiskott-Aldrich syndrome protein,WASp)表达缺失,导致严重疾病(WAS)。错义突变通常导致蛋白表达减少,表现为较轻的 X 连锁血小板减少症(X-linked thrombocytopenia,XLT)或减弱的 WAS [1-3]。然而,最近已经描述了新型的激活突变,导致 X 连锁中性粒细胞减少症(XLN),这是第三种由中性粒细胞减少伴可变骨髓增生不良定义的综合征 [4-6]。WASp 是将信号从细胞膜传递到肌动蛋白细胞骨架的关键,WASp 的缺乏导致细胞骨架缺陷,从而影响正常细胞活动的多个方面,包括增殖、吞噬作用、免疫突触形成、黏附和定向迁移。

相似文献

1
The Wiskott-Aldrich syndrome: The actin cytoskeleton and immune cell function.
Dis Markers. 2010;29(3-4):157-75. doi: 10.3233/DMA-2010-0735.
4
New insights into the biology of Wiskott-Aldrich syndrome (WAS).
Hematology Am Soc Hematol Educ Program. 2009:132-8. doi: 10.1182/asheducation-2009.1.132.
6
WASP: a key immunological multitasker.
Nat Rev Immunol. 2010 Mar;10(3):182-92. doi: 10.1038/nri2724.
7
Wiskott-Aldrich syndrome/X-linked thrombocytopenia in China: Clinical characteristic and genotype-phenotype correlation.
Pediatr Blood Cancer. 2015 Sep;62(9):1601-8. doi: 10.1002/pbc.25559. Epub 2015 Apr 30.
8
Wiskott-Aldrich Syndrome: a model for defective actin reorganization, cell trafficking and synapse formation.
Curr Opin Immunol. 2003 Oct;15(5):585-91. doi: 10.1016/s0952-7915(03)00112-2.
9
WIP remodeling actin behind the scenes: how WIP reshapes immune and other functions.
Int J Mol Sci. 2012;13(6):7629-7647. doi: 10.3390/ijms13067629. Epub 2012 Jun 21.
10
The Wiskott-Aldrich syndrome.
J Allergy Clin Immunol. 2006 Apr;117(4):725-38; quiz 739. doi: 10.1016/j.jaci.2006.02.005.

引用本文的文献

1
First Report of Hematopoietic Stem Cell Transplantation for Children Diagnosed with Wiskott-Aldrich Syndrome in Vietnam.
J Blood Med. 2025 Aug 12;16:373-383. doi: 10.2147/JBM.S528827. eCollection 2025.
2
Inborn Errors of Immunity Presenting with Early-Onset Severe Atopy.
Medicina (Kaunas). 2025 Jan 2;61(1):62. doi: 10.3390/medicina61010062.
3
Expert consensus on the off-label use in China of drugs for rare hematologic diseases (2024 edition).
Front Pharmacol. 2024 Nov 22;15:1477550. doi: 10.3389/fphar.2024.1477550. eCollection 2024.
5
Disulfidptosis, A Novel Cell Death Pathway: Molecular Landscape and Therapeutic Implications.
Aging Dis. 2024 May 2;16(2):917-945. doi: 10.14336/AD.2024.0083.
6
Gene editing-based targeted integration for correction of Wiskott-Aldrich syndrome.
Mol Ther Methods Clin Dev. 2024 Feb 6;32(1):101208. doi: 10.1016/j.omtm.2024.101208. eCollection 2024 Mar 14.
7
Síndrome de Wiskott-Aldrich en España: incidencia, mortalidad y sesgo de género durante 21 años.
Rev Clin Esp. 2023 May;223(5):262-269. doi: 10.1016/j.rce.2023.02.008. Epub 2023 Apr 25.
8
The Wiskott-Aldrich syndrome protein is required for positive selection during T-cell lineage differentiation.
Front Immunol. 2023 Jun 7;14:1188099. doi: 10.3389/fimmu.2023.1188099. eCollection 2023.
9
A Novel Mutation Leading to Wiskott-Aldrich Syndrome in an Ethiopian Boy: a Case Report and a Review of Literature.
J Clin Immunol. 2023 Aug;43(6):1272-1277. doi: 10.1007/s10875-023-01487-7. Epub 2023 Apr 13.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验