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Brief Case Reports
TC is a 28-year-old married man referred by another sleep clinic with a putative diagnosis of narcolepsy and failure to respond to stimulants. (The symptoms reported were severe sleepiness and hypnagogic hallucinations.) Seen with his wife, he described excessive daytime sleepiness dating back to his last years at high school. His sleepiness was dramatic and has led to significant accidents at work and in the home (involving their young children). There were no forensic issues. Mr C had a developmental delay. Interestingly, the patient also reported some of the features of Kleine-Levin syndrome, including dramatically fluctuating appetite and libido and a history of heat stress requiring hospital admission. He had a clear history of night terrors and sleepwalking. His wife described several occasions of sexual initiation while he was asleep leading to full intercourse prior to his awakening. She also described his total disregard for her menstrual status when initiating sexsomnia (very unlike during waking intercourse), and she stated that occasionally in this situation, she would have to shake him to wake him and say "Hey, I've got a pad on." His sleep assessment shows a Periodic Leg Movement Index of 12 per hour and extreme sleepiness on the Multiple Sleep Latency Test (mean sleep latency of 1.9 minutes), even though he was on methylphenidate at the time.
KB, a 37-year-old married police officer, presented at Sleep Rounds in a teaching hospital with severe parasomnia, especially sleepwalking with driving in sleep, particularly when under stress and amplified by alcohol consumption. He had features of upper airways resistance syndrome and apparent response of his parasomnia to clonazepam. At these rounds, he and his wife were asked for the first time about initiation of sexual behavior in sleep during the group interview, to which both responded in the affirmative. A subsequent telephone interview amplified that Mr B would initiate sexual behavior in his sleep approximately once monthly. His wife describes him as more aggressive and more amorous at these times than when he is awake. He indulges in behaviors while asleep that he does not undertake when awake. She says that, in some of these episodes "there is no stopping him"; however, on one occasion when he grabbed her around the neck, she slapped him hard, and he immediately awoke and stopped the behavior. The sleep study showed typical features of NREM parasomnia.
In assessing parasomnic behavior in general and identifying a new disorder in particular, it is necessary to address several important questions regarding complex behavior in sleep. First, one must consider whether it is possible to perform complex acts while asleep. To answer this, we should view the brain as a network of different groups of neurons, which may be variably active. Many subcortical and cortical neurons are indeed inactive during sleep. However, the reticular formation and hippocampal structures, for example, will react even during sleep to any external stimuli, initiating movement to preserve the integrity of the body. On the other hand, cortical structures are normally very active during REM sleep as part of the dream mentation. Also, a good measure of primary and secondary (higher-order) functioning is preserved during sleep, which may give an impression that, during a parasomnic episode, something exists that could be viewed as purposeful act. However, a person experiencing a parasomnic event does not have a fully "awakened" brain — some of the cortical structures, such as those responsible for memorizing and learning or those that help us to distinguish events from objective reality and intrinsic experiences, remain inactive, making some of the higher-order functions, including the consciousness, impaired. As an example, a person with parasomnia can walk, operate a motor vehicle, eat, perform a sexual act, or even kill without the ability to, if we simplify, (fully) control his action. This implies that wakefulness and sleep may occur in a fragmented way and may be concurrent. The result is parasomnic behavior with either complex motor or, as in sexsomnia, motor and autonomic activity (Table 2). At the same time, there is an impairment of consciousness and awareness and, consequently, a relative lack of (legal) responsibility for the resulting behavior.
It is possible that complex (sexual) behavior in sleep is multi-faceted in its etiology. From the neurophysiological perspective, one must consider the possibility of a neurological substrate, such as seizure disorder, brain insult or lesion, toxic reaction (the role of alcohol or psychotropic drug use was evident in several cases we describe), or neurodegenerative disease, as well as genetic inheritance and past physical and sexual abuse. Recent studies prove that some of the complex motor behaviors, namely, episodic nocturnal wanderings, paroxysmal dystonia, and paroxysmal arousals, represent a form of nocturnal seizures with minimal EEG correlates. It is also necessary to stress the relation of sleep apnea, sleep fragmentation, and sleep deprivation to parasomnias. The role of sleep apnea in the case of Mr K is persuasive. Both the resulting hypoxia, but especially the sleep fragmentation leading to partial arousals, may have been factors in triggering the sexsomnia in this case. We also observed features of parasomnia related to SWS and partial arousals in other cases of sexsomnia and made a tentative conclusion that this parasomnia is most likely related to SWS, in degree similar to sleepwalking (with greater margin of confidence, we see it as NREM parasomnia). This would place sexsomnia in the same group with other NREM parasomnias such as sleepwalking, sleep terrors, and confusional arousals, all of which have partial arousal as a main feature, resulting in an intermixed sleep-wake state and complex behavior. From this vantage point, one will appreciate the fact that sexsomnia (with other parasomnias) lies between certain types of sleep-specific seizure disorders on one side of the continuum and dissociative psychiatric disorders on the other. The observation that the sexual behavior in sleep may arise from either a dreamlike experience (or NREM dreaming) or, perhaps, dreaming with sexual content (a feature of rapid eye movement [REM] sleep) is noteworthy. This is not something that would bring the sexsomnia closer to REM parasomnias, since the recall of dreamlike experience has been associated with other types of NREM parasomnias, such as sleep terrors and sleep talking. There is evidence that the organization and affect associated with the sexual behavior during sleep (for example, the cases of DW and KB) is different in sleep, or these behaviors could be a replication of patterns seen during wakefulness (for example, LD).
We consider sexsomnia to be a distinct entity in the family of parasomnias. The unique combination of activated systems in sleep, namely, specific motor and autonomic activation, supports this view. It may be difficult to distinguish between typical sleepwalking and sexsomnia. We propose several guidelines that will assist in this process. The main features of sexsomnia, as opposed to sleepwalking, include frequently present sexual arousal with autonomic activation (for example, erection, vaginal lubrication, ejaculation, sweating, cardiorespiratory response). Sexsomnia without sexual arousal is also reported (for example, in the case of AF), and this may hinder correct diagnosis. The rule of thumb should be to study the behavioral patterns. If there is predominant behavior oriented to the genital areas, there is greater likelihood for sexsomnia, as opposed to parasomnic activity that is only sporadically and incidentally oriented to genital areas. For example, we excluded from this series a patient who was touching his genital area while urinating as part of the more complex range of his sleepwalking behavior. This example would not be equal to touching the genital area in a person in whom this is the primary pattern of behavior and who frequently engages in masturbation in sleep. When assessing the possibility of the sexsomnia in any particular case, it is important not to summarily exclude this condition on a basis of existing sexual arousal. The argument that, in males, nocturnal erections normally do not occur in SWS sleep — thus making it impossible to have a genuine parasomnia, and concurrent erection (sexual arousal) — does not take into account the fact that most parasomnic behavior, with or without sexual content, does not occur in SWS but rather arises out of SWS. Further, it is suggested that the presence of erections implies sexual intent. We were indeed able to establish underlying conscious or subconscious sexual intent in several of our patients with sexsomnia. Sexual intent (particularly subconscious) derived from the sexual drive is deeply rooted in the human psyche. This potent force in human behavior can be recognized in various human activities, and for this reason, we believe that we cannot exclude the possibility of genuine parasomnia that features such underlying intent. When judging the possibility of complex sexual behavior occurring in sleep or out of sleep, one should be cognizant of a range of normal sleep-related phenomena that may or may not have sexual context but do not constitute parasomnic behavior or abnormality in medical terms. These include nocturnal erections, vaginal lubrication, nocturnal emissions, dream orgasms ("wet dreams"), and morning erections (REM sleep related in a postawakening state).
The assessment of patients with sexsomnia should, when possible, include a full EEG as part of the PSG recording with the bed-partner present. Considering the frequency of occurrence, the behavior may not be "caught" in the clinical setting, especially when the number of nights available for PSG recording is limited. The incidence of parasomnic events is generally lower in the clinical setting. This is true for all types of adult parasomnias, and from that point, the sexual behavior in sleep is not an exception.
From the legal perspective "a sleep walker's ability to control voluntarily even complex behavior is severely limited or not available," and it is considered as a cause of "non-insane automatism" (R v Parks). However, the ruling that sleepwalking may, under certain circumstances, constitute a "disease of mind" is also known (R v Burges). The issue of legal responsibility may arise if patients refuse treatment and repeatedly expose themselves to parasomnia-inducing factors and situations, resulting in sexsomnia. All our patients who were legally implicated were subsequently exculpated on a basis of a sane automatism. In these forensic cases, there was evidence of parasomnia in their respective sleep studies and personal history of sleepwalking or sleep talking; further, in absence of any detectable mental illness, neuropsychiatric deficit, or brain disorder, their cases were successful in a court of law. We are not aware of any recidivism, and it appears that these patients do not present a "continuing danger" to society. Finally, when assessing a sexsomnia case, one should always be aware of possible malingering, the incidence of which may be higher than in other parasomnias.
Reflection Exercise #10
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