Katial R K, Lieberman M M, Muehlbauer S L, Lust J A, Hamilos D L
Department of Allergy and Immunology, Walter Reed Army Medical Center, Washington, DC 20307, USA.
J Clin Immunol. 1997 Jan;17(1):34-42. doi: 10.1023/a:1027384311897.
We present the case of a 28-year-old Caucasian female with common variable immunodeficiency (CVID) since age 5 who had a long history of hospitalizations for unexplained fevers and pulmonary infiltrates. The patient developed mild lymphocytosis 7 months prior to our evaluation. Flow cytometry of peripheral blood revealed an expansion of gamma delta T lymphocytes, mild CD4 T lymphocytopenia, and a reduced CD4/CD8 ratio (0.2). Two subpopulations of gamma delta T lymphocytes were found (CD3+/CD4-/CD8+, 47%; CD3+/CD4-/CD8-, 53%), the vast majority of which expressed V-delta 1. An infectious cause for the patient's gamma delta T lymphocytosis could not be found. The sputum was chronically colonized with Staphylococcus aureus, and the organism produced TSST-1 in vitro. A bronchoalveolar lavage (BAL) revealed marked lymphocytosis, but gamma delta T lymphocytes were not overrepresented in the BAL. Lymphocyte functional studies revealed poor proliferative responses to mitogens and staphylococcal superantigens and diminished cytokine production. V-delta 1 T lymphocytes from the patient's blood were not expanded in vitro in response to staphylococcal superantigens. TCR gene rearrangement studies confirmed the presence of J gamma and J beta 1 clonal rearrangements accounting for only a small subpopulation of the gamma delta T lymphocytes. These studies were repeated 5 months later and were unchanged. A bone marrow biopsy was negative for leukemia. Hence, the cause of the patient's gamma delta T lymphocytosis could not be determined despite evaluation for underlying malignancy, occult infection, or superantigen-driven stimulation. The patient ultimately died of progressive respiratory insufficiency. The state of current knowledge regarding gamma delta T lymphocytosis, decreased production of alpha beta T lymphocytes, and a low CD4/ CD8 ratio in association with CVID is discussed.
我们报告一例28岁的白种女性病例,该患者自5岁起患有常见变异型免疫缺陷病(CVID),长期因不明原因发热和肺部浸润而住院。在我们评估前7个月,患者出现轻度淋巴细胞增多。外周血流式细胞术显示γδT淋巴细胞扩增、轻度CD4 T淋巴细胞减少以及CD4/CD8比值降低(0.2)。发现了两个γδT淋巴细胞亚群(CD3+/CD4-/CD8+,47%;CD3+/CD4-/CD8-,53%),其中绝大多数表达V-δ1。未发现该患者γδT淋巴细胞增多的感染性病因。痰液长期被金黄色葡萄球菌定植,且该菌在体外产生毒性休克综合征毒素-1(TSST-1)。支气管肺泡灌洗(BAL)显示明显的淋巴细胞增多,但γδT淋巴细胞在BAL中并未占优势。淋巴细胞功能研究显示对丝裂原和葡萄球菌超抗原的增殖反应较差,细胞因子产生减少。患者血液中的V-δ1 T淋巴细胞对葡萄球菌超抗原在体外未扩增。TCR基因重排研究证实存在Jγ和Jβ1克隆重排,仅占γδT淋巴细胞的一小部分亚群。5个月后重复这些研究,结果未变。骨髓活检白血病为阴性。因此,尽管对潜在恶性肿瘤、隐匿性感染或超抗原驱动的刺激进行了评估,但仍无法确定该患者γδT淋巴细胞增多的原因。患者最终死于进行性呼吸功能不全。本文讨论了目前关于CVID相关的γδT淋巴细胞增多、αβT淋巴细胞产生减少以及低CD4/CD8比值的知识状况。