Department of Neurology, Zhejiang Provincial People's Hospital, People's Hospital of Hangzhou Medical College, Hangzhou, China.
Barrow Neurological Institute, Epilepsy Center, Phoenix Arizona, United States.
Seizure. 2020 Oct;81:186-191. doi: 10.1016/j.seizure.2020.08.018. Epub 2020 Aug 17.
Sleep-related facio-mandibular myoclonus(SRFMM) is a rare and under-recognized stereotyped parasomnia. SRFMM can present with isolated tongue biting, which can be misdiagnosed as epilepsy and sleep bruxism. We aimed to investigate the clinical characteristics and demographics of patients with SRFMM.
We reported a case of SRFMM and presented a literature review on SRFMM. We searched the Medline, Pubmed, and Web of Science database using the following search algorithm: "facio-mandibular myoclonus" or "masticatory myoclonus" or "tongue biting" limited to publications in English.
In total, nine studies were included. In addition to our case, a total of 17 patients were analyzed. SRFMM was found to be more prevalent in males, with a mean age of 48.2 years old. Most of the patients experienced tongue biting during non-rapid eye movement sleep. A majority of the patients were misdiagnosed with epilepsy or sleep bruxism. The simultaneous video EEG and surface EMG was beneficial in confirming the diagnosis of SRFMM. In some patients, clonazepam was reported to ameliorate the tongue biting event.
This study represents a comprehensive summary of SRFMM, which has unique clinical features. Extra-caution may be needed in these cases as it may puzzle neurologists in terms of management.
睡眠相关的面下颌肌阵挛(SRFMM)是一种罕见且未被充分认识的刻板性睡眠障碍。SRFMM 可表现为孤立性舌咬伤,易误诊为癫痫和睡眠磨牙症。本研究旨在探讨 SRFMM 患者的临床特征和人口统计学特征。
我们报告了一例 SRFMM 病例,并对 SRFMM 的文献进行了综述。我们使用以下搜索算法在 Medline、Pubmed 和 Web of Science 数据库中进行搜索:“面下颌肌阵挛”或“咀嚼肌阵挛”或“舌咬伤”,仅限于英文出版物。
共纳入 9 项研究,共分析了 17 例患者。除了我们的病例外,SRFMM 更常见于男性,平均年龄为 48.2 岁。大多数患者在非快速眼动睡眠期间出现舌咬伤。大多数患者被误诊为癫痫或睡眠磨牙症。同时进行视频脑电图和表面肌电图有助于确诊 SRFMM。一些患者报告氯硝西泮可改善舌咬伤事件。
本研究全面总结了 SRFMM 的特征,其具有独特的临床特征。在这些病例中,需要格外小心,因为这可能会使神经科医生在治疗方面感到困惑。