甘氨酸受体自身抗体的结合模式与临床综合征有关。

IF 5.7 2区 医学 Q1 NEUROSCIENCES
Inken Piro, Anna-Lena Wiessler, Eleni Kakavela, Betül Baykan, Erdem Tüzün, Carmen Villmann, Claudia Sommer
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引用次数: 0

摘要

诊断为罕见自身免疫性疾病僵硬人综合征(SPS)或更严重的进行性脑脊髓炎伴僵硬和肌阵挛(PERM)的患者以及脑炎或癫痫患者可能携带针对突触分子的自身抗体,例如甘氨酸受体(GlyR)。这些自身抗体干扰抑制性信号传递,从而引起各种症状。到目前为止,自身抗体相关的潜在病理机制如何导致临床表现的变异性尚不清楚。本研究分析了SPS、PERM和癫痫患者GlyR自身抗体与小鼠中枢神经系统(CNS)组织样本的结合模式,以寻找疾病和患者特异性模式。12例GlyR自身抗体阳性患者按患者初诊情况分组。对这些SPS、PERM和癫痫患者的血清样本进行自身抗体与转染的HEK-293细胞、小鼠脊髓和各种脑组织样本的结合评估。通过与GlyR和突触标记物synaptophysin结合的商业抗体共定位,进一步验证了自身抗体的结合。免疫化学显示,所有患者在转染的HEK-293细胞和小鼠脊髓切片灰质中均有glyr α1特异性自身抗体结合。然而,其他中枢神经系统自身抗体积累增强的区域在SPS、PERM和癫痫患者组之间以及组内存在差异。同样,在每位患者的GlyR表达较高脑区检测到自身抗体沉积。即使标记区域和细胞层之间的差异是患者特异性的,而不是群体特异性的,标记区域的功能与患者受损的功能一致。因此,不同患者的标记区域和细胞层的差异可以解释疾病之间和疾病内部症状的可变性。观察到的多样性表明,有必要采取个性化的方法,将患者特异性自身抗体特性、表型和治疗方法联系起来。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Binding patterns of glycine receptor autoantibodies are related to clinical syndromes.

Patients diagnosed with the rare autoimmune disease Stiff Person Syndrome (SPS) or the more severe form Progressive Encephalomyelitis with Rigidity and Myoclonus (PERM) as well as patients with encephalitis or epilepsy may harbor autoantibodies against synaptic molecules, for example the glycine receptor (GlyR). These autoantibodies interfere with inhibitory signal transmission, which causes a variety of symptoms. How the underlying autoantibody associated pathomechanisms contribute to the variability of clinical presentations, is so far not understood. In this study, binding patterns of GlyR autoantibodies from patients with SPS, PERM and epilepsy to murine central nervous system (CNS) tissue samples were analyzed for disease- and patient-specificity patterns. Twelve GlyR autoantibody positive patients were grouped by the patients' primary diagnoses. Serum samples from these SPS, PERM and epilepsy patients were evaluated for autoantibody binding on transfected HEK-293 cells and murine spinal cord and various brain tissue samples. Autoantibody binding was further verified by co-localization with commercial antibodies binding to the GlyR and the synaptic marker synaptophysin. Immunochemistry revealed GlyRα1-specific autoantibody binding for all included patients on transfected HEK-293 cells and in the grey matter of murine spinal cord sections. Other CNS regions of enhanced autoantibody accumulation however varied among the groups of SPS, PERM and epilepsy patients and also within groups. Similarly, autoantibody deposits were detected in GlyR expressing higher brain areas for each patient. Even if variations between labeled areas and cell layers were rather patient-specific than group-specific, functionality of the labeled areas aligned with the patients' impaired functions. Labeled areas and cell layers differing between patients could thereby explain the variability of symptomatology between and within the diseases. The observed diversity suggests a necessity for a personalized approach correlating patient-specific autoantibody properties, phenotype and treatment approaches.

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来源期刊
Acta Neuropathologica Communications
Acta Neuropathologica Communications Medicine-Pathology and Forensic Medicine
CiteScore
11.20
自引率
2.80%
发文量
162
审稿时长
8 weeks
期刊介绍: "Acta Neuropathologica Communications (ANC)" is a peer-reviewed journal that specializes in the rapid publication of research articles focused on the mechanisms underlying neurological diseases. The journal emphasizes the use of molecular, cellular, and morphological techniques applied to experimental or human tissues to investigate the pathogenesis of neurological disorders. ANC is committed to a fast-track publication process, aiming to publish accepted manuscripts within two months of submission. This expedited timeline is designed to ensure that the latest findings in neuroscience and pathology are disseminated quickly to the scientific community, fostering rapid advancements in the field of neurology and neuroscience. The journal's focus on cutting-edge research and its swift publication schedule make it a valuable resource for researchers, clinicians, and other professionals interested in the study and treatment of neurological conditions.
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