Vermi W, Blanzuoli L, Kraus M D, Grigolato P, Donato F, Loffredo G, Marino C E, Alberti D, Notarangelo L D, Facchetti F
Department of Pathology, Spedali Civili-University of Brescia, Italy.
Am J Surg Pathol. 1999 Feb;23(2):182-91. doi: 10.1097/00000478-199902000-00007.
The Wiskott-Aldrich syndrome (WAS) is a X-linked hematologic disorder characterized by thrombocytopenia, eczema, and immunodeficiency of variable severity. Reported here are the results of a morphologic, morphometric, and immunophenotypic analysis of splenic lymphoid tissue in 12 WAS patients with documented molecular defect and with different disease severity. Spleens from 29 age-matched patients with different diseases were used as controls. Paraffin-embedded tissue (from all cases) and fresh-frozen samples (from 5 WAS patients and 4 control subjects) were used to study the different white pulp compartments by classic morphologic, immunophenotyping, and image analysis techniques. Data were statistically analyzed by both parametric and nonparametric tests. Spleens from WAS patients showed a significant depletion of the total white pulp (p = 0.0008), T cell (p < 0.05), and B cell (p = 0.0002) areas and marginal zone (MZ) thickness (p < 0.0001). Among WAS patients, a negative correlation was found between the score of severity of the disease and all variables considered (Spearman's rank correlation coefficient, r = -0.79, r = -0.73, r = -0.68, and r = -0.56, respectively). In conclusion, this study shows that in WAS a general depletion of the splenic white pulp occurs, supporting the evidence that WAS is characterized by a combined immune defect. The significant reduction of the MZ may explain the inability of WAS patients to mount a response to T-independent antigens.
威斯科特-奥尔德里奇综合征(WAS)是一种X连锁血液系统疾病,其特征为血小板减少、湿疹以及严重程度各异的免疫缺陷。本文报告了对12例经证实存在分子缺陷且疾病严重程度不同的WAS患者脾脏淋巴组织进行形态学、形态计量学和免疫表型分析的结果。将29例年龄匹配的不同疾病患者的脾脏用作对照。采用经典的形态学、免疫表型分析和图像分析技术,利用石蜡包埋组织(所有病例)和新鲜冷冻样本(5例WAS患者和4例对照受试者)研究不同的白髓区室。数据通过参数检验和非参数检验进行统计学分析。WAS患者的脾脏显示白髓总量(p = 0.0008)、T细胞区(p < 0.05)、B细胞区(p = 0.0002)以及边缘区(MZ)厚度(p < 0.0001)均显著减少。在WAS患者中,发现疾病严重程度评分与所有考虑的变量之间呈负相关(斯皮尔曼等级相关系数分别为r = -0.79、r = -0.73、r = -0.68和r = -0.56)。总之,本研究表明,WAS患者脾脏白髓普遍减少,支持了WAS具有联合免疫缺陷这一特征的证据。MZ的显著减少可能解释了WAS患者无法对非T细胞依赖性抗原产生反应的原因。