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老年人中无炎症迹象的抗体相关中枢神经系统综合征

Antibody-associated CNS syndromes without signs of inflammation in the elderly.

作者信息

Escudero Domingo, Guasp Mar, Ariño Helena, Gaig Carles, Martínez-Hernández Eugenia, Dalmau Josep, Graus Francesc

机构信息

From the Service of Neurology (D.E., M.G., C.G., J.D., F.G.), Hospital Clinic, University of Barcelona; Neuroimmunology Program (H.A., C.G., E.M.-H., J.D., F.G.), Institut d'Investigació Biomèdica August Pi i Sunyer; Institució Catalana de Recerca i Estudis Avançats (J.D.), Barcelona, Spain; and Department of Neurology (J.D.), University of Pennsylvania, Philadelphia.

出版信息

Neurology. 2017 Oct 3;89(14):1471-1475. doi: 10.1212/WNL.0000000000004541. Epub 2017 Sep 6.

Abstract

OBJECTIVE

To report the CNS syndromes of patients ≥60 years of age with antibodies against neuronal surface antigens but no evidence of brain MRI and CSF inflammatory changes.

METHODS

This was a retrospective clinical analysis of patients with antibodies against neuronal surface antigens who fulfilled 3 criteria: age ≥60 years, no inflammatory abnormalities in brain MRI, and no CSF pleocytosis. Antibodies were determined with reported techniques.

RESULTS

Among 155 patients ≥60 years of age with neurologic syndromes related to antibodies against neuronal surface antigens, 35 (22.6%) fulfilled the indicated criteria. The median age of these 35 patients was 68 years (range 60-88 years). Clinical manifestations included faciobrachial dystonic seizures (FBDS) in 11 of 35 (31.4%) patients, all with LGI1 antibodies; a combination of gait instability, brainstem dysfunction, and sleep disorder associated with IgLON5 antibodies in 10 (28.6%); acute confusion, memory loss, and behavioral changes suggesting autoimmune encephalitis (AE) in 9 (25.7%; 2 patients with AMPAR, 2 with NMDAR, 2 with GABAbR, 2 with LGI1, and 1 with CASPR2 antibodies); and rapidly progressive cognitive deterioration in 5 (14.3%; 3 patients with IgLON5 antibodies, 1 with chorea; 1 with DPPX antibody-associated cerebellar ataxia and arm rigidity; and 1 with CASPR2 antibodies).

CONCLUSIONS

In patients ≥60 years of age, the correct identification of characteristic CNS syndromes (FBDS, anti-IgLON5 syndrome, AE) should prompt antibody testing even without evidence of inflammation in MRI and CSF studies. Up to 15% of the patients developed rapidly progressive cognitive deterioration, which further complicated the differential diagnosis with a neurodegenerative disorder.

摘要

目的

报告年龄≥60岁、存在抗神经元表面抗原抗体但无脑MRI及脑脊液炎症改变证据的患者的中枢神经系统综合征。

方法

这是一项对符合以下3条标准的抗神经元表面抗原抗体患者的回顾性临床分析:年龄≥60岁、脑MRI无炎症异常、脑脊液无细胞数增多。采用已报道的技术检测抗体。

结果

在155例年龄≥60岁、患有与抗神经元表面抗原抗体相关神经系统综合征的患者中,35例(22.6%)符合指定标准。这35例患者的中位年龄为68岁(范围60 - 88岁)。临床表现包括35例患者中的11例(31.4%)出现面臂肌张力障碍性癫痫(FBDS),均有LGI1抗体;10例(28.6%)出现与IgLON5抗体相关的步态不稳、脑干功能障碍和睡眠障碍;9例(25.7%)出现提示自身免疫性脑炎(AE)的急性意识模糊、记忆力减退和行为改变(2例有AMPAR抗体、2例有NMDAR抗体、2例有GABAbR抗体、2例有LGI1抗体、1例有CASPR2抗体);5例(14.3%)出现快速进展性认知衰退(3例有IgLON5抗体、1例有舞蹈症;1例有DPPX抗体相关的小脑共济失调和手臂僵硬;1例有CASPR2抗体)。

结论

在年龄≥60岁的患者中,即使MRI和脑脊液检查无炎症证据,正确识别特征性中枢神经系统综合征(FBDS、抗IgLON5综合征、AE)也应促使进行抗体检测。高达15%的患者出现快速进展性认知衰退,这使与神经退行性疾病的鉴别诊断更加复杂。

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本文引用的文献

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Clinical manifestations of the anti-IgLON5 disease.抗IgLON5疾病的临床表现。
Neurology. 2017 May 2;88(18):1736-1743. doi: 10.1212/WNL.0000000000003887. Epub 2017 Apr 5.
3
Anti-LGI1 encephalitis: Clinical syndrome and long-term follow-up.抗 LGI1 脑炎:临床综合征和长期随访。
Neurology. 2016 Oct 4;87(14):1449-1456. doi: 10.1212/WNL.0000000000003173. Epub 2016 Sep 2.
4
A clinical approach to diagnosis of autoimmune encephalitis.自身免疫性脑炎的临床诊断方法
Lancet Neurol. 2016 Apr;15(4):391-404. doi: 10.1016/S1474-4422(15)00401-9. Epub 2016 Feb 20.
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Basal ganglia T1 hyperintensity in LGI1-autoantibody faciobrachial dystonic seizures.基底节 T1 高信号在 LGI1 自身抗体面肩肱型肌阵挛癫痫。
Neurol Neuroimmunol Neuroinflamm. 2015 Oct 1;2(6):e161. doi: 10.1212/NXI.0000000000000161. eCollection 2015 Dec.
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Rapidly progressive dementia.快速进展性痴呆
Ann Neurol. 2008 Jul;64(1):97-108. doi: 10.1002/ana.21430.

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