Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, Minnesota, USA.
Hum Immunol. 2010 May;71(5):505-11. doi: 10.1016/j.humimm.2010.02.002. Epub 2010 Mar 12.
Mutations in the TNFRSF13B (TACI) gene have been reported to be associated with Common Variable ImmunoDeficiency (CVID). Of 48 patients evaluated within the immunodeficiency clinic, 39 had CVID, 6 had symptomatic IgA deficiency (IgAD) with or without IgG2 and IgG4 subclass deficiency, while 3 had unclassified immune dysregulatory disorders. In all 48 patients TACI genetic testing was performed, and monoallelic mutations were observed in 4 of the 39 CVID patients (10.26%), an incidence comparable to other studies. Of the 6 IgAD patients, 1 had a heterozygous TACI mutation (16.67%), while of the 3 unclassified patients, 1 had a monoallelic TACI mutation (33.3%), but his sibling who also had the same mutation had CVID. The A181E mutation is one of the statistically significant, among the known TACI gene mutations. Here, 5 of the 6 patients were found to have the A181E mutation (10.42%), which is higher than previously observed. We also evaluated 114 controls and found the A181E mutation in only 1 individual (0.88%). We report in this study that the A181E mutation is associated with a very heterogeneous clinical presentation along with variability in B-cell numbers and amount of TACI protein on memory B cells.
TNFRSF13B(TACI)基因突变与常见可变免疫缺陷(CVID)有关。在免疫缺陷诊所评估的 48 名患者中,39 名患有 CVID,6 名患有有症状的 IgA 缺乏症(IgAD),伴有或不伴有 IgG2 和 IgG4 亚类缺乏症,而 3 名患有未分类的免疫调节障碍疾病。在所有 48 名患者中均进行了 TACI 基因检测,在 39 名 CVID 患者中有 4 名(10.26%)观察到单等位基因突变,发生率与其他研究相当。在 6 名 IgAD 患者中,有 1 名存在杂合 TACI 突变(16.67%),而在 3 名未分类的患者中,有 1 名存在单等位基因突变(33.3%),但也有同样突变的兄弟姐妹患有 CVID。A181E 突变是已知 TACI 基因突变中具有统计学意义的突变之一。在本研究中,6 名患者中有 5 名(10.42%)发现存在 A181E 突变,高于之前的观察结果。我们还评估了 114 名对照者,仅在 1 名个体中发现了 A181E 突变(0.88%)。我们在本研究中报告,A181E 突变与非常异质的临床表现相关,同时伴有 B 细胞数量和记忆 B 细胞上 TACI 蛋白量的变化。