Rachid Rima, Castigli Emanuela, Geha Raif S, Bonilla Francisco A
Division of Immunology, Children's Hospital, Boston,300 Longwood Avenue, Boston, MA 02115, USA.
Curr Allergy Asthma Rep. 2006 Sep;6(5):357-62. doi: 10.1007/s11882-996-0004-9.
Common variable immunodeficiency (CVID) is a heterogeneous primary immunodeficiency disease. Immunoglobulin A deficiency (IGAD) shares some clinical, laboratory, and genetic features with CVID and occurs with relatively greater frequency in first-degree relatives of individuals with CVID. Recently, patients with CVID and IGAD have been found to have mutations of the gene TNFRSF13B encoding the TACI (transmembrane activator and calcium-modulator and cyclophilin-ligand interactor), a member of the tumor necrosis factor-receptor superfamily. In this article, we review the various TACI mutations that have been identified so far. Although six mutations have been reported, no clear genotype-phenotype association has been shown to date. This suggests that the phenotypic expression of TACI mutation is affected by additional genetic and environmental factors. Analysis of a larger sample of patients will be needed to determine if the specific mutations are associated with a particular phenotype or predisposition to the common features of CVID and IGAD: autoimmunity, lymphoproliferation, or malignancy.
普通可变免疫缺陷(CVID)是一种异质性原发性免疫缺陷病。免疫球蛋白A缺乏症(IGAD)与CVID具有一些临床、实验室和遗传特征,并且在CVID患者的一级亲属中出现的频率相对较高。最近,发现CVID和IGAD患者存在编码TACI(跨膜激活剂和钙调节剂以及亲环素配体相互作用分子)的基因TNFRSF13B突变,TACI是肿瘤坏死因子受体超家族的成员。在本文中,我们回顾了迄今为止已鉴定出的各种TACI突变。尽管已报道了六种突变,但迄今为止尚未显示出明确的基因型-表型关联。这表明TACI突变的表型表达受其他遗传和环境因素影响。需要对更大样本的患者进行分析,以确定特定突变是否与特定表型或CVID和IGAD的共同特征(自身免疫、淋巴细胞增殖或恶性肿瘤)的易感性相关。