Violent Sleep-Related Behavior Leading to Subdural Hemorrhage Mark E. Dyken, MD; Deborah C. Lin-Dyken, MD; Peter Seaba, MSEE; Thoru Yamada, MD Objective: To polysomnographically determine, using split-screen electroencephalographic-video analysis, the cause of violent sleep-related activity in a patient whose differential diagnosis includes sleep walking (somnambulism), pavor incubus (adult night terrors), nocturnal seizures, psychogenic wan- dering, and rapid eye movement sleep behavior disorder. Setting: The patient was referred to the University of Iowa, Department of Neurology Sleep Disorders Center, Iowa City, from the local community to The evaluate a history of violent dreams associated with in- jury. The subject presented with a subdural hemorrhage that was discovered with magnetic resonance imaging. Outcome: The diagnosis of rapid eye movement sleep behavior disorder was confirmed after a characteristic spell of violent behavior, with an associated dream, was captured polysomnographically. (Arch Neurol. 1995;52:318-321) rapid eye movement (REM) sleep behavior dis¬ order (RBD) was first de¬ scribed in humans in 1985.1 It is associated with vio¬ lent behavior arising from REM sleep, nor¬ mally a state of essential paralysis. The di¬ agnosis requires a history of potentially harmful sleep-related body movements as¬ sociated with REM dreams.2"5 These be¬ haviors are rarely captured by polysom- nography, as most patients experience a major episode only once every 2 to 3 weeks,6 and, as yet, there exists no spe¬ cific diagnostic criteria based on polysom- nographic findings.5,7 From the Departments of Neurology Sleep Disorders Center (Drs Dyken and Yamada and Mr Seaba) and the Department of Pediatrics, Division of Developmental Disabilities (Dr Lin-Dyken), University of Iowa College of Medicine, Iowa City. After reviewing data from several dif¬ ferent groups, only seven (8.0%) of 87 pa¬ tients with possible RBD reported dreams immediately following characteristic REMrelated behaviors captured polysomno¬ graphically. ·2·4·5·8"1'' Schenck et al20 per¬ formed sleep studies on 85 adult patients with sleep-related injury. Although all RBD patients were reported to be diagnosed polysomnographically, the primary crite¬ rion for diagnosis was loss of REM atonia and emergence of vigorous behaviors dur¬ ing REM sleep. No patients were re¬ ported to have typical, complex behav¬ iors associated with dream mentation captured in the laboratory. Although clinically RBD appears to Downloaded From: http://archneur.jamanetwork.com/ by a University of Iowa User on 06/20/2015 be primarily associated with REM sleep, frequent movements (periodic and nonperiodic) have been noted in non-REM sleep.6 Therefore, polysomnographic sup¬ port for this disorder is usually based pri¬ marily on the observance of elevated muscle tone in REM sleep with nonvio¬ lent movements occurring in both REM and non-REM sleep. Subsequently, many other phenomena associated with el¬ evated muscle tone during REM (narco¬ lepsy,21 drug use,19·22"24 Parkinson's dis¬ ease,25 and the posttraumatic stress disorder26) or excessive movements dur¬ ing sleep (seizure,27 paroxysmal noctur¬ nal dystonia,27"29 somnambulism and night terrors30), psychiatric conditions (such as dissociative states,28 malingering,31 Munchausen syndrome by proxy,31 posttrau¬ matic stress disorder,6 and nocturnal panic attacks6), and sleep drunkenness27·32 (be¬ havioral responses to arousals induced by disorders such as sleep apnea and/or pe¬ riodic limb movements) may be misdiagnosed and inappropriate therapy can occur. REPORT OF A CASE We describe a 73-year-old man with a his¬ tory of tobacco abuse (discontinued after myocardial infarction suffered in 1976). a His only medication was ranitidine hy- Figure 1. Superficial injury sustained to the right maxillary region during a violent sleep behavior. drochloride, 150 mg/d, taken for a remote history of pep¬ tic ulcer disease. He presented with a 10-year history of violent dreams. He stated: "I act them out, sometimes I hurt myself, I go through the motions of what I'm dream¬ ing about." His dreams occurred "almost every night." On occasion, he unintentionally struck his wife. Re¬ Figure 2. Top, A brain magnetic resonance image of the patient revealed a subdural hemorrhage outlined by the black arrows. Bottom, A 6-month follow-up study demonstrates resolution of the hematoma. cently, he dreamed of working on a loading dock and he had seen a man running. He reported: "Someone yelled, 'stop him.' I tried." His wife reported that he jumped off the end of the bed and awoke on the floor with "a bloody lip, head, and knee." During another episode, the patient leaped from his bed, fell, and struck the right side of his face on a corner of a chest (Figure 1 ). He immediately awoke and com¬ plained of a headache and nausea and vomited. He ap¬ peared confused for a 3-hour period, after which he was alert and had no complaints. Results of the neurologic examination were nor¬ mal, and RBD was suspected clinically. A magnetic resonance imaging scan of the brain revealed a leftsided subdural hemorrhage, which had completely resolved by the time of a second study 9 months later (Figure %). A standard polygraph (Nicolet [Madison, Wis] 1A97) used to record the electroencephalogram, electrooculogram, electromyogram (chin, bilateral anterior tibialis, and intercostal muscles), electrocardiogram, and res¬ piratory air flow and effort parameters. The study was recorded and sleep was scored according to the crite¬ rion of Rechtschaffen and Kales." The analog outputs of the 1A97 were input to a reformatter (W/TV-16B, Telefactor Corp, Conshohocken, Pa) that converted the ana¬ log inputs to a video signal, simulating the paper record¬ ing. This simulation was then mixed with a video image of the patient. The combined video image (patient and data) was displayed on a video monitor while being re¬ corded on the video track of a standard videocassette re¬ corder. Audio from the patient's room was recorded on the audio track of the tape. The video image of the pa¬ tient was obtained using a low-level black-and-white video camera and infrared light. The overnight polysomnogram was remarkable for significant periodic limb movements (with a movewas Figure 3. Polysomnographic tracing generally revealed the classic electromyographic augmentation that is associated with rapid eye movement (REM) sleep in the REM sleep behavior disorder. LOG indicates left outer canthus; ROC, right outer canthus; temporal; C, central; ET, ears tied; 0, occipital; LA, left arm; RA, right arm; LL, left leg; RL, right leg; IC, intercostal electromyogram; NA, nasal airflow; OA, oral airflow; and TM, thoracic movement. ment index of 52.0 events per hour and a movement- arousal index of 3.0 events per hour) and an increase in phasic limb movements throughout most REM sleep periods (Figure 3). There was no evidence of sleep apnea or epileptiform activity. Over a 5-month period, a trial of oral clonazepam, 0.5 mg taken orally every evening before sleep, was instituted. The patient reported marked improvement, without complete resolution of his violent nocturnal behavior. Subsequently, clonazepam was discontinued for a 3-day period, after which three consecutive nights of Downloaded From: http://archneur.jamanetwork.com/ by a University of Iowa User on 06/20/2015 Figure 4. These split-screen polysomnogaphic tracings of our patient (left, center, and right) temporally document the course (before, during, and after, respectively) of a typical violent rapid eye movement (REM) behavior followed by a spontaneous dream report. LOG indicates left outer canthus; ROC, right outer canthus; T, temporal; C, central; ET, ears tied; 0, occipital; LA, left arm; RA, right arm; LL, left leg; RL, right leg; IC, intercostal electromyogram; NA, nasal airflow; OA, oral airflow; and TM, thoracic movement. polysomnography were performed. Each study revealed significant periodic limb movements and a general loss of atonia during REM sleep. In the third night study, during one REM episode, after a pro¬ longed period of uncharacteristically quiet electromyographic activity, the patient suddenly exhibited explosive running movements, followed by an arousal (Figure 4). Subsequently, he was coherent and dem¬ onstrated no evidence of confusion. The patient spon¬ taneously reported "one of them dreams. As it started out, I was chasing cattle," after which the animals reportedly turned and pursued him. It should be noted that the patient is a cattle farmer. He was subsequently treated with 1.0 mg of oral clonazepam taken before sleep, with complete resolution of all violent dream activity over the following year. COMMENT appear similar in dream interruption insomnia,36·37 somnambulism/night terrors,27·38·39 seizures, and RBD. Subsequently, RBD has frequently been mlsdiagnosed as nocturnal seizure, psychiatric disorder,27 and sleep apnea.2·6 In addition, there has been a recent pro¬ posal for the existence of "overlapping syndromes" that exhibit characteristics suggestive of both RBD and sleepwalking/night terrors. In the two cases presented by Hurwitz et al,40 complex violent behavior with reported dream imagery was not captured polysomno¬ graphically. Up to 77.1% of patients with RBD present with mildto-moderate sleep-related injuries, including bruises, lac¬ erations, dislocations, and fractures.6 Rarely is the RBD reported to be associated with serious injury, but, ow¬ ing to the limited use of appropriate monitoring tech¬ niques, the true magnitude of these problems may be un¬ derestimated.7·27,28 There has been only one other documented case of associated violent, directed behavior, followed by the RBD causing subdural hematoma.8 A 73-year-old man, with a 2-year history of almost nightly violent movements in sleep, reportedly hit his head on a table "in a dream." A Therefore, few of these patients have polysomno¬ to be associated with dream mentation. Our case is important because we have documented a typical spell of RBD that arises from REM sleep with spontaneous, nonprompted report of a detailed dream that corresponds with observed movements. In the sleep laboratory, videotaped actions, felt to be charac¬ teristic for RBD,are rarely followed by full arousals.4·6·16 computed tomogram of the head revealed bilateral subdu¬ ral hematomas, while polysomnography demonstrated vio¬ lent movements during REM sleep. The nature of the vio¬ lent movements was not described, and none were reported graphically documented violent behavior associated with the report of dreaming, and, to our knowledge, no published studies provide split-screen, videotaped evidence of such a spell, where temporally related behavior and electroencephalographic activity can be appreciated simultaneously. Using advanced split-screen video-imaging tech¬ niques, we were able to capture a classic REM episode sleep other than REM.2·4·26·3435 In addition, violent behaviors associated with dreams have been reported not only in RBD but in nightmares associated with the Accepted for publication November 1, 1993. We acknowledge the comments and technical assis¬ tance given by Harold Adams, MD, and Christine Glenn, REEGT, RPSGT, of the Department of Neurology, Uni¬ versity of Iowa College of Medicine, Iowa City, in the prepa¬ ration of this article. Reprint requests to Department of Neurology, Uni¬ versity of Iowa College of Medicine, Iowa City, IA 52242 (Dr Dyken). Dreamlike mentation can occur from stages of posttraumatic stress disorder,26·35 somnambulism, night terrors, and sleep apnea.27 Schenck et al28 point out that the diagnosis of sleep-related injury cannot be determined only from clinical characteristics of the behavior, the time of night the behavior occurs, or the degree of event or dream recall, as these features can of violent behavior followed by a dream report. These tech¬ nical capabilities allowed us to confidently diagnose and successfully treat RBD in a patient with an associated sub¬ dural hemorrhage. 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