Venzor J, Hua Q, Bressler R B, Miranda C H, Huston D P
Department of Medicine, Baylor College of Medicine, Houston, Texas 77030, USA.
Am J Med Sci. 1997 Apr;313(4):236-8. doi: 10.1097/00000441-199704000-00007.
Herein we report a patient with Behçet's like syndrome, idiopathic CD4+ T-lymphocytopenia, opportunistic infections, and a large polyclonal population of TCR alpha beta + CD4- CD8- T cells. Microfluorimetric analysis of peripheral blood mononuclear cells revealed CD4+ T-cell counts of 10 +/- 5/mm3. The CD3+ T cells were 99% TCR alpha beta +, of which 74 +/- 5% were CD4- CD8-. No clonal populations were detected by southern analysis for T-cell receptor V beta gene rearrangements. No evidence of human immunodeficiency virus infection was present, although nocardia, candida, pneumocystis, cytomegalovirus, and herpes infections were documented. The concomitant presence of opportunistic infections and a large population of TCR alpha beta + CD4- CD8- T cells suggests a pathogenic association and an intense immune response to microbial lipid or lipoglycan antigens presented in the context of CD1 molecules. This case demonstrates the potential for idiopathic CD4+ T-lymphocytopenia to occur in Behçet's-like syndrome with lethal consequences.
在此,我们报告一名患有白塞氏综合征样综合征、特发性CD4 + T淋巴细胞减少症、机会性感染以及大量多克隆TCRαβ + CD4 - CD8 - T细胞群体的患者。对外周血单个核细胞的微量荧光分析显示CD4 + T细胞计数为10±5/mm3。CD3 + T细胞99%为TCRαβ +,其中74±5%为CD4 - CD8 -。通过Southern分析T细胞受体Vβ基因重排未检测到克隆群体。尽管记录了诺卡菌、念珠菌、肺孢子菌、巨细胞病毒和疱疹感染,但未发现人类免疫缺陷病毒感染的证据。机会性感染与大量TCRαβ + CD4 - CD8 - T细胞的同时存在提示了一种致病关联以及对CD1分子背景下呈现的微生物脂质或脂多糖抗原的强烈免疫反应。该病例表明特发性CD4 + T淋巴细胞减少症有可能发生在白塞氏综合征样综合征中并导致致命后果。