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氯巴占成功治疗Heidenhain变异型克雅氏病耐药性癫痫发作:一例报告

Addition of clobazam successfully treating drug resistant seizures in Heidenhain variant Creutzfeldt Jakob disease: A case report.

作者信息

Maille Jason M, Hanna Sebastian S, Shah Darshan N

机构信息

Texas A&M University College of Pharmacy, 59 Reynolds Medical Building, College Station, TX 77843, USA.

University of Vermont Larner College of Medicine, 89 Beaumont Ave, Burlington, VT 05405, USA.

出版信息

Epilepsy Behav Rep. 2023 Jan 12;21:100585. doi: 10.1016/j.ebr.2023.100585. eCollection 2023.

Abstract

Creutzfeldt Jakob Disease (CJD) is a rapidly progressive and fatal neurodegenerative disease that is uncommonly accompanied with seizures. In this case report, we describe a 63-year-old male patient who presented with a 3-week history of visual disturbances and clonic movement of his left arm. Additionally, the patient was reported to have developed erratic behaviors along with insomnia during this period. An EEG showed 4 electrographic seizures of bilateral temporo-occipital onset characterized by 1.5 Hz periodic discharges, lasting 2-13 min. Levetiracetam was started and titrated to the maximal dose however seizures continued so lacosamide and clonazepam were initiated. Despite these aggressive treatments, seizures continued, and oral clobazam 5 mg BID replaced clonazepam. Continued electrographic seizures warranted an increase in clobazam to 10 mg BID after which the seizures stopped; of note, lateralized periodic discharges (LPDs) remained. The patient's symptoms were consistent with the Heidenhain variant, along with probable CJD due to positive RT-QuIC assay, positive 14-3-3 protein, MRI FLAIR hyperintensities, and EEG findings. Although the patient passed away 3 weeks following admission as a result of CJD, we propose that there may be clinical benefit in the use of clobazam in suspected CJD patients presenting with seizures, and its use merits further investigation.

摘要

克雅氏病(CJD)是一种快速进展的致命性神经退行性疾病,很少伴有癫痫发作。在本病例报告中,我们描述了一名63岁男性患者,他有3周的视力障碍和左臂阵挛性运动病史。此外,据报告该患者在此期间还出现了行为异常和失眠。脑电图显示4次双侧颞枕叶起始的电图癫痫发作,特征为1.5赫兹周期性放电,持续2 - 13分钟。开始使用左乙拉西坦并滴定至最大剂量,但癫痫仍持续发作,因此开始使用拉科酰胺和氯硝西泮。尽管采取了这些积极治疗措施,癫痫仍继续发作,口服氯巴占5毫克每日两次替代氯硝西泮。持续的电图癫痫发作促使氯巴占增加至10毫克每日两次,此后癫痫发作停止;值得注意的是,仍存在侧化周期性放电(LPDs)。患者的症状与海登海因变异型相符,同时由于实时震颤诱导转化(RT - QuIC)检测呈阳性、14 - 3 - 3蛋白呈阳性、磁共振成像(MRI)液体衰减反转恢复序列(FLAIR)高信号以及脑电图结果,可能患有克雅氏病。尽管患者入院3周后因克雅氏病去世,但我们认为对于疑似克雅氏病且伴有癫痫发作的患者,使用氯巴占可能具有临床益处,其应用值得进一步研究。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96a6/9867951/2357c6df191f/gr1.jpg

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