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伴有帕金森症和癫痫的额颞叶痴呆与VGKC抗体相关:病例报告及文献综述

Frontotemporal Dementia with Parkinsonism and Epilepsy Associated with VGKC Antibodies: Case Report and Literature Review.

作者信息

Saint Matthew, Alakbarzade Vafa, McLean Brendan

机构信息

College of Medicine and Health, Exeter, United Kingdom.

Department of Neurology, Royal Cornwall Hospitals NHS Trust, Truro, United Kingdom.

出版信息

Case Rep Neurol. 2021 Mar 19;13(1):205-210. doi: 10.1159/000513852. eCollection 2021 Jan-Apr.

DOI:10.1159/000513852
PMID:33976657
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8077438/
Abstract

Antibodies directed against the voltage-gated potassium channel complex (anti-VGKCs) are implicated in several autoimmune conditions including limbic encephalitis and epilepsy. However, emerging evidence suggests that only specific subtypes of anti-VGKCs are pathogenic. We present the case of a 55-year-old man who initially presented with focal unaware seizures and behavioural changes mimicking anti-VGKC-seropositive encephalitis that further progressed to parkinsonism with evidence of frontotemporal dementia and pre-synaptic dopaminergic deficit. Aggressive treatment with immunotherapy was ineffective, and antibody subtyping later revealed the anti-VGKC antibodies to be negative for leucine-rich glioma-associated 1 (LGI1) and contactin-associated protein-like 2 (CASPR2) - the two known pathogenic subtypes. The clinical relevance of so-called "double-negative" anti-VGKCs (i.e., those not directed towards LGI1 or CASPR2) has been called into question in recent years, with evidence to suggest they may be clinically insignificant. Our case emphasises the importance of antibody subtyping in cases of anti-VGKC seropositivity; negative results, particularly when combined with a poor response to immunotherapy, should prompt a rapid reconsideration of the working diagnosis.

摘要

针对电压门控钾通道复合物(抗VGKC)的抗体与包括边缘叶脑炎和癫痫在内的多种自身免疫性疾病有关。然而,新出现的证据表明,只有特定亚型的抗VGKC具有致病性。我们报告了一例55岁男性患者,他最初表现为局灶性无感知发作和行为改变,类似抗VGKC血清阳性脑炎,随后发展为帕金森综合征,伴有额颞叶痴呆和突触前多巴胺能缺陷的证据。积极的免疫治疗无效,抗体亚型分析后来显示抗VGKC抗体对富含亮氨酸的胶质瘤相关蛋白1(LGI1)和接触蛋白相关蛋白样2(CASPR2)这两种已知的致病亚型呈阴性。近年来,所谓的“双阴性”抗VGKC(即不针对LGI1或CASPR2的抗体)的临床相关性受到质疑,有证据表明它们可能在临床上无意义。我们的病例强调了在抗VGKC血清阳性病例中进行抗体亚型分析的重要性;阴性结果,特别是当与免疫治疗反应不佳相结合时,应促使迅速重新考虑初步诊断。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c98a/8077438/1df3953e0644/crn-0013-0205-g02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c98a/8077438/eec071c478f4/crn-0013-0205-g01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c98a/8077438/1df3953e0644/crn-0013-0205-g02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c98a/8077438/eec071c478f4/crn-0013-0205-g01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c98a/8077438/1df3953e0644/crn-0013-0205-g02.jpg

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本文引用的文献

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Expanding spectrum of contactin-associated protein 2 (CASPR2) autoimmunity-syndrome of parkinsonism and ataxia.接触蛋白相关蛋白 2(CASPR2)自身免疫性疾病-帕金森病和共济失调谱的扩展。
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Immunomodulated parkinsonism as a presenting symptom of LGI1 antibody encephalitis.免疫调节性帕金森综合征作为LGI1抗体脑炎的首发症状
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Clinical spectrum and diagnostic value of antibodies against the potassium channel related protein complex.抗钾通道相关蛋白复合物抗体的临床谱及诊断价值
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Treatment of VGKC complex antibody-associated limbic encephalitis: a systematic review.抗电压门控钾通道复合物抗体相关边缘性脑炎的治疗:系统评价。
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