Yeh Shih-Bin, Schenck Carlos H
Department of Neurology (and Sleep Center), Changhua Christian Hospital Yun Lin Branch and Department of Neurology (and Sleep Center), St Martin de Porres Hospital; Chiayi City, Taiwan (ROC).
Minnesota Regional Sleep Disorders Center and Department of Psychiatry, Hennepin County Medical Center and the University of Minnesota Medical School, Minneapolis, MN, USA.
Sleep Sci. 2014 Sep;7(3):170-7. doi: 10.1016/j.slsci.2014.09.016. Epub 2014 Oct 5.
To present findings on a series of cases of sporadic nocturnal frontal lobe epilepsy (NFLE), a form of NFLE that is infrequently reported, in contrast to familial (autosomal dominant) NFLE. Both forms of NFLE need to be distinguished from parasomnias, nocturnal temporal lobe epilepsy, and other nocturnal disorders.
Eight consecutive cases of sporadic NFLE were evaluated at a sleep clinic in Taiwan. All patients had clinical evaluations, daytime waking and sleeping EEGs, brain MRIs, and overnight video-polysomnography (vPSG) with seizure montage.
Gender was equal (four males, four females); mean age was 18.4 yrs (range, 7-41 yrs). Age of NFLE onset was by puberty. Premorbid history was negative for any neurologic, medical or psychiatric disorder. NFLE subtypes: nocturnal paroxysmal dystonia, n=6; paroxysmal arousals, n=2. MRI brain scan abnormalities with clinical correlates were found in one patient. Daytime awake EEGs were negative for ictal/interictal activity in all patients, but two patients had daytime sleep EEGs with interictal epileptiform EEG activity. During vPSG studies, three of eight patients with NFLE seizure events had concurrent epileptiform EEG activity, and two patients had interictal epileptiform EEG activity during their vPSG studies. No case had a spontaneous remission. Anticonvulsant therapy was highly effective in all eight cases (>75% reduction in seizure frequency).
These cases confirm that sporadic NFLE closely resembles familial NFLE, and comprises a set of distinct clinical manifestations, with variable intensity, and variable scalp EEG epileptiform abnormalities across sleep and wakefulness, which have previously been identified in Caucasian patients from Europe and North America.
报告一系列散发性夜间额叶癫痫(NFLE)病例的研究结果,散发性NFLE是NFLE的一种形式,与家族性(常染色体显性)NFLE相比,其报道较少。两种形式的NFLE均需与异态睡眠、夜间颞叶癫痫及其他夜间疾病相鉴别。
在台湾一家睡眠诊所对8例连续的散发性NFLE病例进行评估。所有患者均接受了临床评估、白天清醒及睡眠脑电图检查、脑部磁共振成像(MRI)以及带有发作监测的夜间视频多导睡眠图(vPSG)检查。
性别均衡(4例男性,4例女性);平均年龄为18.4岁(范围7 - 41岁)。NFLE发病年龄在青春期。病前无任何神经、医学或精神疾病史。NFLE亚型:夜间阵发性肌张力障碍,n = 6;阵发性觉醒,n = 2。1例患者脑部MRI扫描异常且与临床相关。所有患者白天清醒脑电图均未发现发作期/发作间期活动,但2例患者白天睡眠脑电图有发作间期癫痫样脑电图活动。在vPSG研究期间,8例NFLE发作事件患者中有3例同时出现癫痫样脑电图活动,2例患者在vPSG研究期间有发作间期癫痫样脑电图活动。无一例自发缓解。抗惊厥治疗对所有8例患者均非常有效(发作频率降低>75%)。
这些病例证实散发性NFLE与家族性NFLE极为相似,具有一系列独特的临床表现,强度各异,且在睡眠和清醒状态下头皮脑电图癫痫样异常情况也有所不同,此前在欧洲和北美的白种患者中已有发现。