MacCarthy-Morrogh L, Gaspar H B, Wang Y C, Katz F, Thompson L, Layton M, Jones A M, Kinnon C
Molecular Immunology Unit, The Institute of Child Health, London, United Kingdom.
Clin Immunol Immunopathol. 1998 Jul;88(1):22-7. doi: 10.1006/clin.1998.4557.
Wiskott-Aldrich syndrome (WAS) is an X-linked primary immunodeficiency that is usually associated with thrombocytopenia and eczema. The very variable phenotype of WAS results from defects in the WAS protein (WASP), the function of which is not well understood. In many cases causative mutations have now been identified in the WAS gene. Attempts have been made to correlate the nature of the mutations with the severity of the disease. In this study we investigated mutations in 13 patients with WAS and analyzed the expression of WASP in patient blood samples by immunoblot analysis. We found that despite extensive variation in the nature of the mutations in patients with severe WAS symptoms, none express the protein. However, in 1 patient with a mild clinical phenotype WASP expression was detected. Such an analysis could be used as an initial screening procedure for the diagnosis of WAS prior to genotypic analysis.
威斯科特-奥尔德里奇综合征(WAS)是一种X连锁原发性免疫缺陷病,通常与血小板减少症和湿疹相关。WAS的表型变化很大,是由威斯科特-奥尔德里奇综合征蛋白(WASP)缺陷引起的,其功能尚不清楚。目前已在许多病例中鉴定出WAS基因的致病突变。人们试图将突变的性质与疾病的严重程度联系起来。在本研究中,我们调查了13例WAS患者的突变情况,并通过免疫印迹分析患者血样中WASP的表达。我们发现,尽管重症WAS症状患者的突变性质存在广泛差异,但均未表达该蛋白。然而,在1例临床表型较轻的患者中检测到了WASP表达。这种分析可作为在进行基因分型分析之前诊断WAS的初步筛查程序。