Takeuchi Yosuke, Masuda Teruaki, Matsuta Hiroyuki, Yabuuchi Kenichi, Hata Nobuhiro, Fujiki Minoru, Obayashi Konen, Matsubara Etsuro, Kimura Noriyuki
Department of Neurology, Faculty of Medicine, Oita University, Oita, Japan.
Department of Neurosurgery, Faculty of Medicine, Oita University, Oita, Japan.
Epilepsy Behav Rep. 2025 Apr 22;30:100773. doi: 10.1016/j.ebr.2025.100773. eCollection 2025 Jun.
Ictal cold shiver is a rare manifestation of seizures that originate in the temporal lobes; it is characterized by body trembling and sensations of cold. This study aims to investigate the clinical manifestations and etiological factors in patients with ictal cold shiver. We reviewed 59 cases of ictal cold shiver by using various keywords to search published original articles and case reports, and we described a new case of autoimmune limbic encephalitis. The median age at diagnosis was higher in patients with autoimmune encephalitis than in those with other etiologies (p = 0.007). Autoimmune encephalitis showed more frequent bilateral temporal lobe abnormalities on magnetic resonance imaging (MRI) (p = 0.027) and generalized or bilateral independent epileptiform discharges on electroencephalography (EEG) (p = 0.047). Anti-leucine-rich glioma-inactivated 1 antibodies were most commonly detected in patients with autoimmune encephalitis with ictal cold shiver (85.7 %). This study highlights the importance of considering autoimmune mechanisms in the presence of ictal cold shiver, especially in patients with bilateral temporal lobe lesions on MRI, generalized or bilateral independent epileptiform discharges on EEG, and older onset.
发作期冷颤是起源于颞叶的癫痫发作的一种罕见表现；其特征为身体颤抖和寒冷感。本研究旨在调查发作期冷颤患者的临床表现和病因。我们通过使用各种关键词检索已发表的原始文章和病例报告，回顾了59例发作期冷颤病例，并描述了1例自身免疫性边缘叶脑炎的新病例。自身免疫性脑炎患者的诊断中位年龄高于其他病因患者（p = 0.007）。自身免疫性脑炎在磁共振成像（MRI）上显示双侧颞叶异常更为频繁（p = 0.027），在脑电图（EEG）上显示广泛性或双侧独立癫痫样放电更为频繁（p = 0.047）。在伴有发作期冷颤的自身免疫性脑炎患者中，最常检测到抗富含亮氨酸胶质瘤失活1抗体（85.7%）。本研究强调了在发作期冷颤存在时考虑自身免疫机制的重要性，尤其是在MRI显示双侧颞叶病变、EEG显示广泛性或双侧独立癫痫样放电且起病年龄较大的患者中。
