Matthews Ian, Sims Gary, Ledwidge Serena, Stott David, Beeson David, Willcox Nick, Vincent Angela
Neurosciences Group, Weatherall Institute of Molecular Medicine, John Radcliffe Hospital, Oxford OX3 9DS, United Kingdom.
Lab Invest. 2002 Oct;82(10):1407-17. doi: 10.1097/01.lab.0000032379.63784.9c.
The weakness in myasthenia gravis (MG) is mediated by autoantibodies against adult muscle acetylcholine receptors (AChR) at the neuromuscular junction; most of these antibodies also bind to fetal AChR, which is present in the thymus. In rare cases, babies of mothers with MG, or even of asymptomatic mothers, develop a severe developmental condition, arthrogryposis multiplex congenita, caused by antibodies that inhibit the ion channel function of the fetal AChR while not affecting the adult AChR. Here we show that these fetal AChR inhibitory antibodies are significantly more common in females sampled after pregnancy than in those who present before pregnancy, suggesting that they may be induced by the fetus. Moreover, we were able to clone high-affinity combinatorial Fab antibodies from thymic cells of two mothers with MG who had babies with arthrogryposis multiplex congenita. These Fabs were highly specific for fetal AChR and did not bind the main immunogenic region that is common to fetal and adult AChR. The Fabs show strong biases to VH3 heavy chains and to a single Vkappa1 light chain in one mother. Nevertheless, they each show extensive intraclonal diversification from a highly mutated consensus sequence, consistent with antigen-driven selection in successive steps. Collectively, our results suggest that, in some cases of MG, initial immunization against fetal AChR is followed by diversification and expansion of B cells in the thymus; maternal autoimmunity will result if the immune response spreads to the main immunogenic region and other epitopes common to fetal and adult AChR.
重症肌无力(MG)的肌无力由针对神经肌肉接头处成人肌肉乙酰胆碱受体(AChR)的自身抗体介导;这些抗体中的大多数也与胸腺中存在的胎儿AChR结合。在极少数情况下,患有MG的母亲甚至无症状母亲的婴儿会患上一种严重的发育疾病,即先天性多发性关节挛缩症,这是由抑制胎儿AChR离子通道功能而不影响成人AChR的抗体引起的。我们在此表明,这些胎儿AChR抑制性抗体在产后采样的女性中比孕前采样的女性中更为常见,这表明它们可能是由胎儿诱导产生的。此外,我们能够从两名患有先天性多发性关节挛缩症婴儿的MG母亲的胸腺细胞中克隆出高亲和力组合Fab抗体。这些Fab对胎儿AChR具有高度特异性,不与胎儿和成人AChR共有的主要免疫原性区域结合。在一名母亲中,这些Fab对VH3重链和单个Vκ1轻链表现出强烈偏好。然而,它们各自都显示出从高度突变的共有序列开始的广泛克隆内多样化,这与连续步骤中的抗原驱动选择一致。总体而言,我们的结果表明,在某些MG病例中,针对胎儿AChR的初始免疫接种之后是胸腺中B细胞的多样化和扩增;如果免疫反应扩散到胎儿和成人AChR共有的主要免疫原性区域和其他表位,就会导致母体自身免疫。