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Wiskott-Aldrich 综合征生物学的新见解。

New insights into the biology of Wiskott-Aldrich syndrome (WAS).

机构信息

Institute of Child Health, London, United Kingdom.

出版信息

Hematology Am Soc Hematol Educ Program. 2009:132-8. doi: 10.1182/asheducation-2009.1.132.

Abstract

The Wiskott-Aldrich syndrome (WAS) is an X-linked immunodeficiency disease with a characteristic clinical phenotype that includes thrombocytopenia with small platelets, eczema, recurrent infections due to immunodeficiency, and an increased incidence of autoimmune manifestations and malignancies. The identification of the molecular defect in the WAS gene has broadened the clinical spectrum of disease to include chronic or intermittent X-linked thrombocytopenia (XLT), a relatively mild form of WAS, and X-linked neutropenia (XLN) due to an arrest of myelopoiesis. The pathophysiological mechanisms relate to defective actin polymerization in hematopoietic cells as a result of deficient or dysregulated activity of the WAS protein (WASp). The severity of disease is variable and somewhat predictable from genotype. Treatment strategies therefore range from conservative through to early definitive intervention by using allogeneic hematopoietic stem cell transplantation and potentially somatic gene therapy. All aspects of the condition from clinical presentation to molecular pathology and basic cellular mechanisms have been reviewed recently.

摘要

威斯科特-奥尔德里奇综合征(WAS)是一种 X 连锁免疫缺陷病,具有特征性的临床表型,包括血小板减少伴小血小板、湿疹、免疫缺陷引起的反复感染,以及自身免疫表现和恶性肿瘤的发生率增加。WAS 基因的分子缺陷的确定拓宽了疾病的临床谱,包括慢性或间歇性 X 连锁血小板减少症(XLT),一种相对较轻的 WAS 形式,以及由于髓系造血停滞引起的 X 连锁中性粒细胞减少症(XLN)。病理生理机制与造血细胞中肌动蛋白聚合的缺陷有关,这是由于 WAS 蛋白(WASp)的活性不足或失调所致。疾病的严重程度从基因型上看是可变的,并且具有一定的可预测性。因此,治疗策略从保守治疗到早期通过异基因造血干细胞移植和潜在的体基因治疗进行明确干预。从临床表现到分子病理学和基础细胞机制的疾病的各个方面最近都得到了回顾。

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