Department of Neurology, Kitasato University School of Medicine, Sagamihara, Japan.
Front Immunol. 2023 Jan 16;13:1066830. doi: 10.3389/fimmu.2022.1066830. eCollection 2022.
A variety of neuronal surface (NS) antibodies (NS-Ab) have been identified in autoimmune encephalitis (AE). Tissue-based assay (TBA) using a rodent brain immunohistochemistry (IHC) is used to screen NS-Ab, while cell-based assay (CBA) to determine NS antigens. Commercial rat brain IHC is currently available but its clinical relevance remains unclear. Immunostaining patterns of NS antigens have not been extensively studied yet. To address these issues, we assessed a predictive value of "neuropil pattern" and "GFAP pattern" on commercial IHC in 261 patients, and characterized an immunostaining pattern of 7 NS antigens (NMDAR, LGI1, GABAaR, GABAbR, AMPAR, Caspr2, GluK2). Sensitivity and specificity of "neuropil pattern" for predicting NS-Ab were 66.0% (95% CI 55.7-75.3), and 98.2% (95% CI 94.8-99.6), respectively. False-positive rate was 1.8% (3/164) while false-negative rate was 34.0% (33/97). In all 3 false-positive patients, neuropil-like staining was attributed to high titers of GAD65-Ab. In 33 false-negative patients, NMDAR was most frequently identified (n=18 [54.5%], 16/18 [88.9%] had low titers [< 1:32]), followed by GABAaR (n=5). Of 261 patients, 25 (9.6%) had either GFAP (n=21) or GFAP-mimicking pattern (n=4). GFAP-Ab were identified in 21 of 31 patients examined with CBA (20 with GFAP pattern, 1 with GFAP-mimicking pattern). Immunostaining pattern of each NS antigen was as follows: 1) NMDAR revealed homogenous reactivity in the dentate gyrus molecular layer (DG-ML) with less intense dot-like reactivity in the cerebellar granular layer (CB-GL); 2) both GABAaR and GluK2 revealed intense dot-like reactivity in the CB-GL, but GABAaR revealed homogenous reactivity in the DG-ML while GluK2 revealed intense reactivity along the inner layer of the DG-ML; and 3) LGI1, Caspr2, GABAbR, and AMPAR revealed intense reactivity in the cerebellar ML (CB-ML) but LGI1 revealed intense reactivity along the middle layer of the DG-ML. Whereas, Caspr2, GABAbR, and AMPAR revealed similar reactivity in the DG-ML but some difference in other regions. TBA is useful not only for screening NS- or GFAP-Ab but also for estimating NS antigens; however, negative results should be interpreted cautiously because "neuropil pattern" may be missed on commercial IHC when antibody titers are low. Antigen-specific immunoreactivity is a useful biomarker of AE.
自身免疫性脑炎 (AE) 中已鉴定出多种神经元表面 (NS) 抗体 (NS-Ab)。使用啮齿动物脑免疫组织化学 (IHC) 的基于组织的检测 (TBA) 用于筛选 NS-Ab,而基于细胞的检测 (CBA) 用于确定 NS 抗原。目前可获得商业性大鼠脑 IHC,但临床相关性尚不清楚。NS 抗原的免疫染色模式尚未得到广泛研究。为了解决这些问题,我们在 261 名患者中评估了商业 IHC 中“神经胶质模式”和“GFAP 模式”的预测价值,并对 7 种 NS 抗原 (NMDAR、LGI1、GABAaR、GABAbR、AMPA、Caspr2、GluK2) 的免疫染色模式进行了特征描述。“神经胶质模式”预测 NS-Ab 的敏感性和特异性分别为 66.0%(95%CI 55.7-75.3)和 98.2%(95%CI 94.8-99.6)。假阳性率为 1.8%(3/164),而假阴性率为 34.0%(33/97)。在所有 3 例假阳性患者中,神经胶质样染色归因于 GAD65-Ab 的高滴度。在 33 例假阴性患者中,NMDAR 最常被识别 (n=18 [54.5%],16/18 [88.9%] 的滴度较低 [<1:32]),其次是 GABAaR (n=5)。在 261 名患者中,25 名 (9.6%) 患者具有 GFAP(21 名)或 GFAP 模拟模式 (4 名)。在 31 名接受 CBA 检查的患者中检测到 21 例 GFAP-Ab(20 例具有 GFAP 模式,1 例具有 GFAP 模拟模式)。每个 NS 抗原的免疫染色模式如下:1)NMDAR 在齿状回分子层 (DG-ML) 中显示均匀反应性,在小脑颗粒层 (CB-GL) 中显示点状反应性较弱;2)GABAaR 和 GluK2 在 CB-GL 中均显示强烈的点状反应性,但 GABAaR 在 DG-ML 中显示均匀反应性,而 GluK2 沿 DG-ML 的内层显示强烈反应性;3)LGI1、Caspr2、GABAbR 和 AMPAR 在小脑 ML (CB-ML) 中显示强烈反应性,但 LGI1 在 DG-ML 的中层显示强烈反应性。然而,Caspr2、GABAbR 和 AMPAR 在 DG-ML 中显示相似的反应性,但在其他区域有一些差异。TBA 不仅可用于筛选 NS 或 GFAP-Ab,还可用于估计 NS 抗原;然而,当抗体滴度较低时,商业 IHC 可能会错过“神经胶质模式”,因此应谨慎解释阴性结果。抗原特异性免疫反应是 AE 的有用生物标志物。