Felix Sandra, Thobois Stephane, Peter-Derex Laure
Unité d'hypnologie, Service de Neurologie fonctionnelle et d'épileptologie, Hospices civils de Lyon, Hôpital Neurologique, Bron, France.
Université Claude Bernard Lyon 1, Lyon, France.
J Sleep Res. 2016 Apr;25(2):211-5. doi: 10.1111/jsr.12364. Epub 2016 Jan 18.
A 75-year-old man complained of excessive daytime sleepiness (EDS), difficulty falling asleep and nocturnal agitation during sleep. Restless legs syndrome (RLS) was diagnosed and treated. Because of persistent EDS, snoring and nycturia, a nocturnal polysomnography (PSG) was performed. PSG showed high sleep fragmentation related to a moderate to severe obstructive sleep apnea syndrome. Continuous positive airway pressure treatment (CPAP) was proposed. Because of the persistence of abnormal nocturnal behaviours, characterized by screaming, punching and falling out of bed, a video-PSG with CPAP treatment was performed. The recording showed typical chin electromyography (EMG) activity increase associated with violent movements during rapid eye movement (REM) sleep, suggesting REM sleep behaviour disorders (RBD). Clinical neurological examination found no parkinsonian syndrome, no dysautonomic sign and no neurological focal sign. Dopamine transporter imaging [123I-FP-CIT single photon emission computed tomography (SPECT)] did not find any presynaptic dopaminergic pathways degeneration. Brain magnetic resonance imaging showed a vascular lesion suggestive of cavernoma located in the pons. The present case illustrates the complexity of sleep disturbance diagnosis with a possible entanglement of aetiologies responsible for nocturnal agitation, and confirms that an isolated pons cavernoma should be considered among the rare causes of RBD.
一名75岁男性主诉白天过度嗜睡(EDS)、入睡困难及夜间睡眠时烦躁不安。诊断为不安腿综合征(RLS)并进行了治疗。由于持续存在EDS、打鼾及夜尿症,遂进行了夜间多导睡眠图(PSG)检查。PSG显示睡眠片段化严重,与中度至重度阻塞性睡眠呼吸暂停综合征相关。建议进行持续气道正压通气治疗(CPAP)。由于夜间异常行为持续存在,表现为尖叫、拳打脚踢及从床上跌落,遂进行了CPAP治疗下的视频PSG检查。记录显示在快速眼动(REM)睡眠期间,典型的颏肌电图(EMG)活动增加并伴有暴力动作,提示REM睡眠行为障碍(RBD)。临床神经学检查未发现帕金森综合征、自主神经功能障碍体征及神经局灶性体征。多巴胺转运体成像[123I-FP-CIT单光子发射计算机断层扫描(SPECT)]未发现任何突触前多巴胺能通路退变。脑磁共振成像显示桥脑有一个提示海绵状血管瘤的血管病变。本病例说明了睡眠障碍诊断的复杂性,可能存在导致夜间烦躁不安的病因相互交织的情况,并证实孤立的桥脑海绵状血管瘤应被视为RBD的罕见病因之一。