Department of Immunology, Oxford Radcliffe Hospitals, Oxford, UK.
Clin Exp Immunol. 2010 Sep;161(3):536-41. doi: 10.1111/j.1365-2249.2010.04216.x.
Common variable immunodeficiency disorders (CVIDs) are a heterogeneous group of diseases characterized by hypogammaglobulinaemia and consequent susceptibility to infection. CVID patients commonly develop a variety of additional manifestations for which the causative factors are not fully understood. Two such manifestations are granulomatous disease and enteropathy. Because the ability to predict complications would aid clinical management, we continue to search for possible disease modifier genes. NOD2 acts a microbial sensor and is involved in proinflammatory signalling. Particular mutations of the NOD2 gene are associated with Crohn's disease including gly908arg, leu1007finsc and arg702trp polymorphisms. We hypothesized that NOD2 polymorphisms may be a disease modifier gene towards an enteropathic or granulomatous phenotype within CVIDs. Sequence-specific primers returned genotypes for 285 CVID patients from centres across the United Kingdom and Europe. We present the frequencies of the different phenotypes of patients within our international cohort. Arg702trp polymorphisms were significantly less frequent than wild-type (WT) (P = 0·038) among international CVID patients with splenomegaly. Gly908arg polymorphisms were more prevalent than WT in UK patients with autoimmune disorders (P = 0·049) or enteropathy (P = 0·049). NOD2 polymorphisms were not more prevalent than WT in CVID patients with clinical phenotypes of granulomata. UK allele frequencies of 0·014, 0·056 and 0·026 were found for gly908arg, arg702trp and leu1007finsc NOD2 polymorphisms, respectively. These do not differ significantly from UK immunocompetent controls confirming, as expected, that in addition these NOD2 polymorphisms do not confer susceptibility to CVIDs per se.
普通变异性免疫缺陷病(CVID)是一组异质性疾病,其特征为低丙种球蛋白血症和随之而来的感染易感性。CVID 患者通常会出现多种其他表现,其致病因素尚未完全了解。其中两种表现为肉芽肿病和肠病。由于预测并发症的能力将有助于临床管理,我们继续寻找可能的疾病修饰基因。NOD2 作为微生物传感器,参与促炎信号转导。NOD2 基因的特定突变与包括 gly908arg、leu1007finsc 和 arg702trp 多态性在内的克罗恩病有关。我们假设 NOD2 多态性可能是 CVID 中肠病或肉芽肿表型的疾病修饰基因。来自英国和欧洲各地中心的 285 名 CVID 患者的序列特异性引物返回了基因型。我们展示了我们国际队列中患者不同表型的频率。Arg702trp 多态性在伴有脾肿大的国际 CVID 患者中明显低于野生型(WT)(P = 0·038)。Gly908arg 多态性在伴有自身免疫性疾病(P = 0·049)或肠病(P = 0·049)的英国患者中比 WT 更为常见。NOD2 多态性在具有肉芽肿临床表型的 CVID 患者中并不比 WT 更为常见。在英国人群中,gly908arg、arg702trp 和 leu1007finsc NOD2 多态性的等位基因频率分别为 0·014、0·056 和 0·026。这些与英国免疫功能正常的对照没有显著差异,这证实了,正如预期的那样,这些 NOD2 多态性本身并不能赋予 CVID 易感性。