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小儿波瓦桑脑炎病例系列中运动障碍现象学的谱与演变

Spectrum and Evolution of Movement Disorder Phenomenology in a Pediatric Powassan Encephalitis Case Series.

作者信息

Yang Kathryn, Lindsay Rebecca, Quiroz Vicente, Srouji Rasha, Ebrahimi-Fakhari Darius

机构信息

Movement Disorders Program, Department of Neurology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts, USA.

Department of Pediatrics, Section of Neurology, Cumming School of Medicine, University of Calgary, Calgary, Alberta, Canada.

出版信息

Mov Disord Clin Pract. 2024 Dec;11(12):1613-1619. doi: 10.1002/mdc3.14245. Epub 2024 Oct 22.

Abstract

BACKGROUND

The Powassan virus is a rare neurotropic, tick-borne arbovirus associated with meningoencephalitis. Despite the virus's known predilection for the basal ganglia, there are no reports detailing the spectrum of movement disorders in children with Powassan meningoencephalitis.

CASES

We present 3 cases of pediatric Powassan encephalitis, highlighting the diverse and evolving movement disorders associated with this disease. We observed subcortical myoclonus and progressive generalized dystonia (patient 1), transient dyskinesias and refractory focal dystonia (patient 2), and generalized dystonia evolving into chorea and lingual dyskinesias (patient 3). One patient exhibited multifocal vasculitis on magnetic resonance imaging angiography, a novel finding.

CONCLUSIONS

Movement disorders were a primary source of the morbidity experienced by pediatric Powassan encephalitis patients throughout their disease course, underscoring the importance of regular monitoring and adaptable treatment strategies in this condition. Larger, prospective studies are necessary to fully delineate the spectrum of associated movement disorders in this rare and severe disease.

摘要

背景

波瓦桑病毒是一种罕见的嗜神经性、蜱传虫媒病毒,与脑膜脑炎相关。尽管已知该病毒易侵犯基底神经节,但尚无关于波瓦桑脑膜脑炎患儿运动障碍谱的详细报道。

病例

我们报告3例儿童波瓦桑脑炎病例,突出了与该疾病相关的多样且不断演变的运动障碍。我们观察到皮质下肌阵挛和进行性全身性肌张力障碍(患者1)、短暂性运动障碍和难治性局灶性肌张力障碍(患者2),以及全身性肌张力障碍演变为舞蹈症和舌运动障碍(患者3)。一名患者在磁共振血管造影上表现为多灶性血管炎,这是一个新发现。

结论

运动障碍是儿童波瓦桑脑炎患者整个病程中发病的主要原因,强调了在这种情况下定期监测和适应性治疗策略的重要性。需要开展更大规模的前瞻性研究,以全面描绘这种罕见且严重疾病相关运动障碍的谱。

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