Andersen Pernille, Permin Henrik, Andersen Vagn, Schejbel Lone, Garred Peter, Svejgaard Arne, Barington Torben
Department of Clinical Immunology, Blood Bank, University Hospital, Copenhagen, Copenhagen, Denmark.
Blood. 2005 Jan 15;105(2):511-7. doi: 10.1182/blood-2003-12-4359. Epub 2004 Sep 14.
Reduced levels of somatic hypermutation (SHM) have recently been described in IgG-switched immunoglobulin genes in a minority of patients with common variable immunodeficiency (CVID), demonstrating a disruption of the normal linkage between isotype switch and SHM. To see if, irrespective of isotype, there is a tendency to use unmutated immunoglobulin genes in CVID, we studied SHM in kappa light-chain transcripts using a VkappaA27-specific restriction enzyme-based hot-spot mutation assay (IgkappaREHMA). Hot-spot mutations were found in 48% (median; reference interval, 28%-62%) of transcripts from 53 healthy controls. Values were significantly lower in 31 patients (median, 7.5%; range, 0%-73%; P < .0000001) of whom 24 (77%) had levels below the reference interval. Low levels of SHM correlated with increased frequency of severe respiratory tract infection (SRTI; P < .005), but not with diarrhea (P = .8). Mannose-binding lectin (MBL) deficiency also correlated with SRTI score (P = .009). However, the correlation of SHM and SRTI was also seen when only patients with normal MBL genotypes were analyzed (n = 18, P = .006). A slight decline of mutated fractions over years was noted (P = .01). This suggests that most patients with CVID fail to recruit affinity-maturated B cells, adding a qualitative deficiency to the quantitative deficiency characterizing these patients.
最近在少数常见可变免疫缺陷(CVID)患者的IgG类别转换免疫球蛋白基因中发现体细胞超突变(SHM)水平降低,这表明同种型转换与SHM之间的正常联系被破坏。为了了解在CVID中是否存在不考虑同种型而使用未突变免疫球蛋白基因的倾向,我们使用基于VκA27特异性限制酶的热点突变分析(IgκREHMA)研究了κ轻链转录本中的SHM。在53名健康对照的转录本中,48%(中位数;参考区间,28%-62%)发现了热点突变。31例患者的值显著降低(中位数,7.5%;范围,0%-73%;P <.0000001),其中24例(77%)低于参考区间。低水平的SHM与严重呼吸道感染(SRTI)频率增加相关(P <.005),但与腹泻无关(P =.8)。甘露糖结合凝集素(MBL)缺乏也与SRTI评分相关(P =.009)。然而,仅分析MBL基因型正常的患者时(n = 18,P =.006),也观察到SHM与SRTI之间的相关性。注意到多年来突变分数略有下降(P =.01)。这表明大多数CVID患者未能募集亲和力成熟的B细胞,这些患者在定量缺陷的基础上又增加了定性缺陷。