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日本脑炎病毒感染中的上下运动神经元混合损伤

Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection.

作者信息

Ghosh Ritwik, Dubey Souvik, Chatterjee Subhankar, Kanti Ray Biman, Benito-León Julián

机构信息

Department of General Medicine, Burdwan Medical College and Hospital, Burdwan, India.

Department of Neuromedicine, Bangur institute of Neurosciences, Kolkata, India.

出版信息

Case Rep Neurol. 2020 Dec 18;12(3):482-488. doi: 10.1159/000510711. eCollection 2020 Sep-Dec.

Abstract

Cerebral manifestations in Japanese B encephalitis are well known. However, there are very few studies focusing on extra-cerebral manifestations, among which focal anterior horn cell involvement is exceedingly rare. We herein report a case of Japanese B encephalitis with focal anterior horn cell involvement and unfurl how stepwise clinical approach and targeted investigations helped to solve the diagnostic conundrum. A 27-year-old female was admitted with fever, headache, altered sensorium, and convulsions. She tested positive for Japanese B encephalitis-IgM. Following conservative management, she regained consciousness after 5 days when neurological examination revealed marked cognitive impairment, medial convergence of eyeballs, upward gaze restriction, upper limbs dystonia with brisk tendon jerks, and flaccid paraparesis. A repeat neurological examination, on day 15 of admission, showed marked wasting and intermittent fasciculation in both lower limbs. Brain magnetic resonance imaging showed asymmetrical (right > left) bilateral thalamic and midbrain lesions, hyperintense on T2 and T2-fluid-attenuated inversion recovery (FLAIR)-weighted imaging with mild diffusion restriction on diffusion-weighted imaging and apparent diffusion coefficient map, suggestive of encephalitis. Nerve conduction study revealed decreased compound muscle action potentials exclusively in lower limbs with intact sensory nerve action potentials. Electromyogram showed chronic denervation potentials and presence of spontaneous activity in lower limbs, but not in upper limbs, indicative of focal anterior horn cell involvement. Prognosis of Japanese B encephalitis does not only depend on cerebral sequelae. Anterior horn cell involvement can dictate poor outcome and can easily be missed if not carefully dealt with.

摘要

日本乙型脑炎的脑部表现众所周知。然而,针对脑外表现的研究非常少,其中局灶性前角细胞受累极为罕见。我们在此报告一例伴有局灶性前角细胞受累的日本乙型脑炎病例,并阐述逐步的临床方法和针对性检查如何有助于解决诊断难题。一名27岁女性因发热、头痛、意识改变和惊厥入院。她的日本乙型脑炎IgM检测呈阳性。经过保守治疗,5天后她恢复了意识,此时神经检查发现明显的认知障碍、眼球内聚、向上凝视受限、上肢肌张力障碍伴腱反射亢进以及弛缓性截瘫。入院第15天的再次神经检查显示双下肢明显消瘦和间歇性肌束震颤。脑部磁共振成像显示双侧丘脑和中脑不对称性(右侧>左侧)病变,在T2加权成像和T2液体衰减反转恢复(FLAIR)加权成像上呈高信号,在扩散加权成像和表观扩散系数图上有轻度扩散受限,提示脑炎。神经传导研究显示仅下肢复合肌肉动作电位降低,感觉神经动作电位正常。肌电图显示下肢存在慢性失神经电位和自发活动,但上肢没有,提示局灶性前角细胞受累。日本乙型脑炎的预后不仅取决于脑部后遗症。前角细胞受累可能导致不良预后,如果处理不当很容易被漏诊。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bbf9/7841720/46bb85858bb2/crn-0012-0482-g01.jpg

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