Department of Neurosurgery, Center of Epilepsy, Beijing Institute for Brain Disorders, Beijing Key Laboratory of Epilepsy Research, Sanbo Brain Hospital, Capital Medical University, Beijing, 100093, China.
Department of Brian Institute, Center of Epilepsy, Beijing Institute for Brain Disorders, Beijing Key Laboratory of Epilepsy Research, Sanbo Brain Hospital, Capital Medical University, Beijing, 100093, China.
Brain Pathol. 2020 Mar;30(2):246-260. doi: 10.1111/bpa.12770. Epub 2019 Aug 22.
Rasmussen encephalitis (RE) is a severe pediatric inflammatory brain disease characterized by unilateral inflammation and atrophy of the cerebral cortex, drug-resistant focal epilepsy and progressive neurological and cognitive deterioration. The etiology and pathogenesis of RE remain unclear. Our previous results demonstrated that the adenosine A1 receptor (A1R) and the major adenosine-removing enzyme adenosine kinase play an important role in the etiology of RE. Because the downstream pathways of inhibitory A1R signaling are modulated by stimulatory A2AR signaling, which by itself controls neuro-inflammation, glial activation and glial glutamate homeostasis through interaction with glutamate transporter GLT-1, we hypothesized that maladaptive changes in adenosine A2A receptor (A2AR) expression are associated with RE. We used immunohistochemistry and Western blot analysis to examine the expression of A2ARs, glutamate transporter-I (GLT-1) and the apoptotic marker Bcl-2 in surgically resected cortical specimens from RE patients (n = 18) in comparison with control cortical tissue. In lesions of the RE specimen we found upregulation of A2ARs, downregulation of GLT-1 and increased apoptosis of both neurons and astroglia. Double staining revealed colocalization of A2ARs and Bcl-2 in RE lesions. These results suggest that maladaptive changes in A2AR expression are associated with a decrease in GLT-I expression as a possible precipitator for apoptotic cell loss in RE. Because A2AR antagonists are already under clinical evaluation for Parkinson's disease, the A2AR might likewise be a tractable target for the treatment of RE.
拉森姆脑炎(RE)是一种严重的儿童炎症性脑病,其特征为单侧大脑皮质炎症和萎缩、耐药性局灶性癫痫以及进行性神经和认知功能恶化。RE 的病因和发病机制尚不清楚。我们之前的研究结果表明,腺苷 A1 受体(A1R)和主要的腺苷清除酶腺苷激酶在 RE 的发病机制中起着重要作用。由于抑制性 A1R 信号的下游途径受刺激性 A2AR 信号的调节,而 A2AR 信号本身通过与谷氨酸转运体 GLT-1 相互作用来控制神经炎症、胶质细胞激活和胶质细胞谷氨酸稳态,我们假设腺苷 A2A 受体(A2AR)表达的适应性变化与 RE 有关。我们使用免疫组织化学和 Western blot 分析来检测手术切除的 RE 患者(n=18)皮质标本中 A2ARs、谷氨酸转运体-I(GLT-1)和凋亡标志物 Bcl-2 的表达情况,并与对照皮质组织进行比较。在 RE 标本的病变中,我们发现 A2ARs 的上调、GLT-1 的下调以及神经元和星形胶质细胞的凋亡增加。双重染色显示 A2ARs 和 Bcl-2 在 RE 病变中存在共定位。这些结果表明,A2AR 表达的适应性变化与 GLT-I 表达的减少有关,这可能是 RE 中凋亡细胞丢失的一个促成因素。由于 A2AR 拮抗剂已经在帕金森病的临床评估中,A2AR 也可能成为治疗 RE 的可行靶点。