Sexsomnia has been reported and is well described in 115 prior cases in the literature. There have been associations with other sleep disorders serving as triggers for confusional arousals, thereby worsening sexsomnia episodes. We present a case of an adolescent boy with a history of resected and treated pineoblastoma who later developed sexsomnia marked by multiple episodes of masturbatory events per night. He had additional suspicions of obstructive sleep apnea. Polysomnography confirmed severe obstructive sleep apnea and captured multiple episodes of sexsomnia from both REM and NREM sleep. The patient also had daytime symptoms of severe anxiety and hypersomnia that required pharmacological intervention, cognitive behavioral techniques, and hypnosis. The patient showed improvement with hypnosis along with a multimodal approach to the treatment of sexsomnia.
Contreras JB, Richardson J, Kotagal S. Sexsomnia in an adolescent. J Clin Sleep Med. 2019;15(3):505–507.
Sleep-related abnormal sexual behaviors are classified by the third edition of the International Classification of Sleep Disorders as a subtype of disorders of arousal.1 These behaviors are a result of confusional arousals out of NREM sleep that may manifest as masturbation, coital-like pelvic movements, fondling of a partner, sexual vocalizations, intercourse, or orgasm with no recall of the episode. Medico-legal implications have been described, leading to charges of sexual assault, rape, child molestation—all related to automatic and amnestic behaviors performed while asleep.2 Patients frequently have psychological symptoms of anxiety, depression, guilt, and shame, leading some to consider suicide.3 Though sexsomnia is generally reported in adults, we present a case of a teenager with this condition and discuss its pathophysiology and relevance to clinicians caring for adolescents.
REPORT OF CASE
A 16-year old male presented to our sleep clinic for the evaluation of excessive daytime sleepiness, snoring, and sexual behaviors during sleep. His father had observed that the patient masturbated multiple times a night while seemingly asleep. The patient's sleep had become increasingly fragmented and his daytime anxiety worsened. He worried that he might inadvertently carry out these behaviors at his friends' houses, hence began to decline invitations for sleep overs.
His sleep history was significant for loud snoring, snort arousals, unrefreshing sleep, intermittent dry mouth, and fatigue upon awakening in the morning. He was sleepy in the daytime, with a score of 22/32 on the Pediatric Daytime Sleepiness Scale (reference value is 16 or less).4 The excessive daytime sleepiness was compounded by a recent increase of nighttime lorazepam dose to 6 mg for anxiety by his psychiatrist. He had gained 30 pounds in weight over the preceding year. There was also a history of dream enactment behavior with flailing and shouting during dreams. He had occasionally punched holes through his bedroom wall during these episodes.
The patient had a past medical history of a pineoblastoma resection 4 years prior to the sleep evaluation. He subsequently underwent craniospinal radiation, chemotherapy with vincristine, lomustine, cisplatinum and cyclophosphamide. A follow-up head MRI 3 years after diagnosis showed a resolution of prior brain lesions. He developed panhypopituitarism for which hormone replacement was provided with daily hydrocortisone, levothyroxine, and testosterone. There was concern that testosterone may have precipitated masturbation, so the dose was reduced, without improvement. Following the brain tumor resection, he had also developed attention deficit hyperactivity disorder, mood disorder, and anxiety.
The patient underwent nocturnal polysomnography with time-synchronized video and audio recording, along with 16-derivation electroencephalogram, arm and leg electromyogram derivations, and the usual cardiopulmonary monitoring. A total of 356 minutes of sleep were recorded with sleep efficiency of 76%, all sleep stages were represented. An apneahypopnea index of 15 events/h was noted, consisting of supine and non-supine obstructive apneas and hypopneas, classified as severe obstructive sleep apnea (OSA) based on the pediatric scoring criteria of the American Academy of Sleep Medicine. Additionally, the patient had four episodes of stereotypic masturbatory behaviors during the initial sleep study, each occurring after arousals triggered by obstructive apnea events out of REM sleep (Figure 1). There was preservation of the physiologic REM sleep atonia. Repeat polysomnography was performed, showing successful control of sleep-disordered breathing with continuous positive airway pressure (CPAP) of 8 cmH2O and four masturbatory events out of NREM sleep. Lorazepam was discontinued given its lack of benefit. He was prescribed CPAP. Follow-up after 2 months showed excellent CPAP adherence, average nightly use of 5 hours and 36 minutes at a pressure of 7 cmH2O, and a residual apnea-hypopnea index of 2.6 events/h. There was also a modest reduction in the frequency of sleep-related sexual behaviors and improved daytime alertness (based upon history provided by the patient and his parents) during this visit. Given the persistence of sleep fragmentation, he was prescribed clonazepam 0.5 mg at bed time. It was subsequently discontinued due to increased daytime sleepiness. Follow-up 6 months later showed re-emergence of excessive daytime sleepiness and increased frequency of nocturnal sexsomnia behaviors despite excellent CPAP adherence. The parents also reported automatic daytime behaviors such as “taking off driving” in the middle of the day, frequently without destination. There had also been an episode of severe anxiety and suicidality that required hospitalization. There was a concern of a bidirectional relationship between daytime sleepiness and anxiety/depression. Consequently, a Multiple Sleep Latency Test (MSLT) was performed to evaluate for hypersomnia. It showed an unequivocal mean sleep latency of 7.63 minutes, without sleep-onset REM periods, consistent with hypersomnia (reference value > 10–12 minutes for teenagers). He was started on modafinil to enhance daytime alertness.
Obstructive sleep apnea event out of REM sleep that triggered an arousal and a masturbatory event.
Obstructive sleep apnea event out of REM sleep that triggered an arousal and a masturbatory event.
While on modafinil, there was improvement in his overall levels of alertness, thought process and symptoms of anxiety. Further management included psychological counseling and several sessions of hypnosis for sexsomnia. The hypnosis treatment was tried empirically by one of the co-authors (JR) given prior success on long-term follow-up in patients with various parasomnias.5,6 At follow-up a year later, he reported significantly reduced frequency of nocturnal sexual events, had lost a significant amount of weight, and reported overall improvements in levels of anxiety.
Though 115 cases of sexsomnia have been reported in the literature,3,7–9 the condition is likely underreported.1,3,7,9 Sexsomnia generally manifests itself in males, with disease onset between 26 and 63 years of age.3,7–9 An internet survey noted that symptom onset may be earlier in patients with a history of sleepwalking and also involve more females than initially suspected.10 Our case is unusual in that it manifested itself in a 16-year-old man with a history of pineoblastoma resection. Only three cases have been reported in the literature involving teenage patients that presented with symptoms of fondling and masturbation.3,7 The early age of onset, and the possible bidirectional relationship between OSA and parasomnias on the one hand and anxiety/depression on the other hand made management of this complex nocturnal behavior more challenging. Also, our patient did not have complete resolution of sexomnia after sleep-disordered breathing treatment as previously described,9,11 requiring a multimodal approach to treatment.
Our case is only the second case of sexsomnia with behaviors captured during polysomnography in an adolescent or young adult.12 The prior case involved a 20-year-old soldier with prior history of childhood sleepwalking who exhibited reactivated sleepwalking and new-onset sexsomnia after starting military shift work. Episodes of masturbation were captured during polysomnography that occurred during stage N2 and N3 sleep with associated confusional arousals not related to respiratory events. He did not show REM sleep behavior sleep disorder or REM sleep without atonia, though these have been previously reported in patients with sexsomnia.12
We feel that sleep fragmentation was multifactorial, as there was some persistence of masturbatory events despite adequate titration during polysomnography. Most reports have shown significant improvement in episodes of sexsomnia in patients receiving treatment for comorbid OSA.3,8 In our case, treatment of OSA with CPAP led to a modest improvement over time, supported by a prior report of persistent symptoms despite treatment with CPAP for OSA.11 Inadequate response in our case was likely due to associated anxiety. Residual hypersomnia related to OSA likely also hindered the patient from benefiting from psychological counseling. Multiple therapeutic modalities were ultimately needed to reduce the frequency of the sleep-related sexual behaviors, including treatment of OSA with CPAP, daytime modafinil for daytime sleepiness, bedtime clonazepam, psychological counseling, and hypnosis. Automatic behaviors may have been related to daytime sleepiness, though we cannot be certain of this. Hypnosis proved to be an effective therapeutic modality as it resulted in the greatest improvement of symptoms in our refractory case. Hypnosis is hypothesized to lead to a state of deep physical relaxation with retention of a focused mind so that new thoughts can be introduced.5,6 In a prospective study of 36 patients with various parasomnias (with mean duration of parasomnias of 18.7 years), patients reported spell-free or “much improved” response rates of 54% at 18 months and 50% at 5 years following one to two hypnosis sessions.5 Sleepwalkers fared better with overall improvement of 67% at 5 years. A retrospective study of hypnotherapy of NREM parasomnias found similar benefit.6 One advantage in choosing hypnotherapy over cognitive behavioral therapy (CBT) was that hypnotherapy only requires 1–6 sessions whereas CBT may require a longer period of exposure. In refractory cases of parasomnias such as ours, hypnosis could be a viable adjunctive option in the management of parasomnias. The dream-enactment behaviors seen at night were most likely a form of pseudo-RBD related to sleep fragmentation from OSA.13
Given the rarity of the condition, no systematic pharmacological trials have been performed for patients with sexsomnia. Prior cases have anecdotally shown good response to pharmacological therapy including clonazepam, fluoxetine, trimipramine, lamotrigine, paroxetine, escitalopram and duloxetine.3,7–9 The most experience exist with clonazepam as a method to reduce confusional arousals. Our patient unfortunately did not tolerate treatment.
Our case highlights the complex clinical presentation of sexsomnia in adolescents. It also underscores need for a team-based management approach that involves the primary care provider, child and adolescent psychiatrist, child psychologist, and sleep specialist.
All authors listed have reviewed and approved this manuscript. Off-label or investigational use: sodium oxybate and clomipramine. The authors report no conflicts of interest.