Molecular Immunology Unit, UCL Institute of Child Health, London, UK.
Dis Markers. 2010;29(3-4):157-75. doi: 10.3233/DMA-2010-0735.
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.
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。