Hyperventilation-induced cerebellar fit in a patient with Chiari 1.5 malformation

IF 1.9 4区 医学 Q3 CLINICAL NEUROLOGY
Ryota Sasaki, Masako Kinoshita, Nahomi Osugi, Hidehiro Hirabayashi, Ichiro Nakagawa
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Abstract

Cerebellar fit (CF) is characterized by paroxysmal head retroflexion and opisthotonus with preserved consciousness.1-3 Here, we present a case of a 43-year-old male with cough syncope (CS) due to Chiari 1.5 malformation, who exhibited CF during hyperventilation (HV).4 EEG revealed a normal posterior dominant rhythm of 8–9 Hz. Approximately 150 s after initiating HV, the patient experienced truncal convulsion accompanied by opisthotonic posturing (Video 1). Notably, the patient retained his consciousness with open eyes. No electrographic seizure patterns were evident, the posterior dominant rhythm was preserved, and the electrocardiogram demonstrated normal sinus rhythm (Figure 1). He underwent a foramen magnum decompression and his CS and CF disappeared. As a mechanism, HV may induce dissociation between intracranial and spinal cord cerebrospinal fluid pressures, which further lower the cerebellum and compress the brainstem and cerebellum.2-5 CF may be caused by the Guillain–Mollaret triangle disruption and CS by dysfunction of the brainstem reticular formation.6, 7

This study was partially supported by JPJSBP 120217720 and by the Nakatani Foundation for Advancement of Measuring Technologies in Biomedical Engineering : Technology Exchange Program.

None of the authors have any conflicts of interest to declare.

Abstract Image

一名恰里氏 1.5畸形患者过度换气引起的小脑瘫痪。
小脑强直(CF)的特征是阵发性头部后屈和肌张力不全,但意识保持清醒。1-3 在此,我们介绍了一例因Chiari 1.5畸形导致咳嗽性晕厥(CS)的 43 岁男性患者,他在过度通气(HV)时表现出小脑强直。启动 HV 约 150 秒后,患者出现截肢抽搐,并伴有瞳孔散大的姿势(视频 1)。值得注意的是,患者睁眼时仍保持清醒。没有明显的电图癫痫发作模式,后占位心律得以保留,心电图显示窦性心律正常(图 1)。他接受了枕骨大孔减压术,CS和CF消失了。2-5 CF 可能由 Guillain-Mollaret 三角区破坏引起,CS 则由脑干网状结构功能障碍引起、7 本研究得到 JPJSBP 120217720 和 Nakatani Foundation for Advancement of Measuring Technologies in Biomedical Engineering : Technology Exchange Program 的部分支持。
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来源期刊
Epileptic Disorders
Epileptic Disorders 医学-临床神经学
CiteScore
4.10
自引率
8.70%
发文量
138
审稿时长
6-12 weeks
期刊介绍: Epileptic Disorders is the leading forum where all experts and medical studentswho wish to improve their understanding of epilepsy and related disorders can share practical experiences surrounding diagnosis and care, natural history, and management of seizures. Epileptic Disorders is the official E-journal of the International League Against Epilepsy for educational communication. As the journal celebrates its 20th anniversary, it will now be available only as an online version. Its mission is to create educational links between epileptologists and other health professionals in clinical practice and scientists or physicians in research-based institutions. This change is accompanied by an increase in the number of issues per year, from 4 to 6, to ensure regular diffusion of recently published material (high quality Review and Seminar in Epileptology papers; Original Research articles or Case reports of educational value; MultiMedia Teaching Material), to serve the global medical community that cares for those affected by epilepsy.
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