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恙虫病相关的眼阵挛:印度队列中的临床病程及纵向结局

Scrub Typhus-Associated Opsoclonus: Clinical Course and Longitudinal Outcomes in an Indian Cohort.

作者信息

Ralph Ravikar, Prabhakar A T, Sathyendra Sowmya, Carey Ronald, Jude John, Varghese George M

机构信息

Department of Internal Medicine, Christian Medical College Hospital, Vellore, Tamil Nadu, India.

Department of Neurology, Christian Medical College Hospital, Vellore, Tamil Nadu, India.

出版信息

Ann Indian Acad Neurol. 2019 Apr-Jun;22(2):153-158. doi: 10.4103/aian.AIAN_198_18.

DOI:10.4103/aian.AIAN_198_18
PMID:31007425
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6472253/
Abstract

CONTEXT

Opsoclonus, a rare neurological manifestation in scrub typhus, causes significant distress and disability. There is a paucity of clinical data and outcomes in these patients.

AIM

This study aims to describe the clinical and laboratory profile and longitudinal outcomes in a scrub typhus patient cohort with opsoclonus.

SETTINGS AND DESIGN

This retrospective study was conducted in a 2700-bed teaching hospital in South India, in scrub typhus patients with opsoclonus over a 5-year period.

PATIENTS AND METHODS

Clinical, laboratory, and radiological data and outcomes at discharge and 6- and 12-weeks postdischarge were documented.

RESULTS

Of 1650 scrub typhus patients, 18 had opsoclonus. 17 had opsoclonus at presentation, while one patient developed opsoclonus on the 5 admission day, 1-day postdefervescence. Opsoclonus was first noted after a median interval of 11 (7-18) days from fever onset. It was associated with myoclonus in 94% (17/18), cerebellar dysfunction in 67% (12/18), extrapyramidal syndrome (EPS) in 33% (6/18), and aseptic meningitis in 17% (3/18) patients. Mean cerebrospinal fluid (CSF) white blood cell (WBC) count was 9 ± 2.7 cells/cumm, with mean CSF protein 118.5 ± 53.9 mg% and mean CSF glucose 97 ± 13 mg% in 1l/15 patients. Brain magnetic resonance imaging was unremarkable in 75% (9/12). Case-fatality rate was 5.5% (1/18). Complete resolution of the index neurological syndrome occurred at 12-week postdischarge.

CONCLUSIONS

Opsoclonus is a rare neurological manifestation in scrub typhus, usually occurring in association with myoclonus, cerebellar dysfunction, or EPS. It appears to occur during the resolving febrile phase, with neurological deficits completely resolving at 12 weeks.

摘要

背景

眼阵挛是恙虫病一种罕见的神经学表现,会导致严重不适和功能障碍。这些患者的临床数据和预后情况较少。

目的

本研究旨在描述一组患有眼阵挛的恙虫病患者的临床和实验室特征以及长期预后。

设置与设计

这项回顾性研究在印度南部一家拥有2700张床位的教学医院进行,研究对象为5年间患有眼阵挛的恙虫病患者。

患者与方法

记录临床、实验室和放射学数据以及出院时、出院后6周和12周时的预后情况。

结果

在1650例恙虫病患者中,18例有眼阵挛。17例在就诊时就有眼阵挛,而1例患者在入院第5天、退热后1天出现眼阵挛。眼阵挛首次出现的中位间隔时间为发热开始后11(7 - 18)天。94%(17/18)的患者伴有肌阵挛,67%(12/18)伴有小脑功能障碍,33%(6/18)伴有锥体外系综合征(EPS),17%(3/18)的患者伴有无菌性脑膜炎。15例患者中有11例的平均脑脊液(CSF)白细胞(WBC)计数为9 ± 2.7个/立方毫米,平均脑脊液蛋白为118.5 ± 53.9毫克%,平均脑脊液葡萄糖为97 ± 13毫克%。75%(9/12)的患者脑部磁共振成像无明显异常。病死率为5.5%(1/18)。出院后12周时,首发神经综合征完全缓解。

结论

眼阵挛是恙虫病一种罕见的神经学表现,通常与肌阵挛、小脑功能障碍或EPS相关。它似乎发生在热退期,神经功能缺损在12周时完全缓解。

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Autops Case Rep. 2014 Mar 31;4(1):29-37. doi: 10.4322/acr.2014.005. eCollection 2014 Jan-Mar.
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Neuroblastoma presenting as opsoclonus-myoclonus: A series of six cases and review of literature.以眼阵挛-肌阵挛为表现的神经母细胞瘤:6例病例系列及文献复习
J Pediatr Neurosci. 2016 Oct-Dec;11(4):373-377. doi: 10.4103/1817-1745.199462.
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Cerebellitis as an atypical manifestation of scrub typhus.
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