A challenging case of sexsomnia in an adolescent female presenting with depressive-like symptoms

Abstract

Sleep-related sexualized behaviors occur in the parasomnia known as sexsomnia, recognized as a variant of confusional arousals in the International Classification of Sleep Disorders, third edition. These instinctive behaviors of a sexual nature emerge from deep non-rapid eye movement sleep, and patients often present with distinguishing features within this sleep disorder category. There are often adverse psychosocial consequences and not uncommonly medicolegal implications. While associations to psychiatric consequences from the sexsomnia have been demonstrated and efforts to further typify this condition have been made, sexsomnia remains incompletely characterized in the more than 200 published cases to date, with male predominance. We now present the first reported case of an adolescent female with sexsomnia that was triggered by the onset of Crohn’s disease and its treatment with azathioprine and with interpersonal consequences leading to an initial psychiatric consultation on account of depressive symptoms. These symptoms were deemed to be secondary to the sexsomnia. In addition to describing unusual and clinically relevant features in this case of sexsomnia, this original case provides insights into triggers, predisposing factors, perpetuating factors, and therapeutic considerations that are important for raising awareness in sleep clinicians, primary care providers, and mental health professionals.

Citation:

Brás J, Schenck CH, Andrade R, et al. A challenging case of sexsomnia in an adolescent female presenting with depressive-like symptoms. J Clin Sleep Med. 2023;19(10):1845–1847.

INTRODUCTION

Sleep-related abnormal sexual behaviors or sexsomnia/sleepsex recurrent sexual behaviors that typically occur during an incomplete non-rapid eye movement (NREM) sleep awakening with subsequent amnesia.^1–3 Behaviors range from sexual vocalizations, orgasms, sexualized movements, and masturbation to full sexual intercourse.^1,3–6 Usually, these behaviors are related to confusional arousals but may also occur in sleepwalking, although less frequently.^1,7,8 The first classification of sexsomnia was published in 2007.⁶

Sexsomnia often arises in individuals with other NREM sleep parasomnias, such as confusional arousals, sleepwalking, or sleep-related eating disorder, with sexsomnia often being the last parasomnia to emerge.^3–7 Sexual behaviors can also be observed with confusional awakenings caused by respiratory sleep disorders, namely obstructive sleep apnea.^4,5,7 Similarly, there are also cases described with rapid eye movement (REM) sleep parasomnias (ie, parasomnia overlap disorder), circadian rhythm disorders, nocturnal seizures, restless legs syndrome, sleep-related bruxism, periodic limb movement disorder, and narcolepsy.^5,7 Stress, fatigue, sleep deprivation, drug or alcohol use, and close physical contact with the bed partner have also been reported as precipitating factors.^3–8 Clonazepam is the most widely used drug in the treatment of sexsomnia; however, there are reported cases of successful treatment with other pharmacological agents and with therapies controlling comorbid obstructive sleep apnea.^2,5–7

We report a case of sexsomnia in an adolescent female that has unusual and noteworthy features.

REPORT OF CASE

We present the case of an 18-year-old female, body mass index of 19.7 kg/m², who initially presented for psychiatric consultation on account of depressive symptoms in association with complaints of disturbed sleep with sexual behaviors. The presenting symptoms of sadness, emotional lability, and embarrassment were strictly related to the sexsomnia and the negative impact on the patient’s life. The psychiatric consultation identified these symptoms as being secondary to the sexsomnia, without any underlying psychopathology being identified. After the absence of any history of depression or any other psychiatric disorder was confirmed, she was referred to a psychiatrist with expertise in sleep disorders. She had a previous history of sleepwalking in childhood, with no recurrent episodes since early adolescence. She reported the onset of sleep-related sexual behaviors 6 months before the psychiatric consultation, shortly after she had started azathioprine therapy for newly diagnosed Crohn’s disease. Upon arising in the morning, she experienced pubic pain, without realizing the cause of this symptom. However, after sleeping with her boyfriend, he told her about the new onset of recurrent masturbatory behaviors during sleep. These episodes occurred 1 to 2 times a night, shortly after falling asleep. During the episodes there was no involvement with the partner. There was amnesia for these sexsomnia episodes, and no associated dreams were reported. Upon being informed of these episodes, she felt ashamed, sad, and worried about her relationship, since her partner did not understand the reason for her sleep masturbation, as their sexual activity was described as regular and satisfactory and without any problematic issues. Moreover, there was no history of traumatic sexual experiences.

The patient reported an irregular sleep schedule with reduced total sleep time, sometimes sleeping less than 6 hours. When questioned, she denied any symptoms of restless legs syndrome, snoring, apneas, daytime sleepiness, or insomnia. Furthermore, the depression diagnosis was discarded. Instead, some embarrassment and worries still prevailed in the specific context of the sleep-related sexual behaviors.

Current medications included azathioprine and melatonin in a dose of 1 mg at bedtime, which was initiated right after the initial assessment. There was a positive family history for parasomnia, involving the mother with a history of sleepwalking.

At the sleep consultation, the Pittsburgh Sleep Quality Index of 7/21 validated the perception of a poor sleep quality, and the STOP-Bang questionnaire revealed a low risk for obstructive sleep apnea.

A nocturnal video polysomnography was performed that revealed a decrease in sleep efficiency (66.8%) and fragmented sleep with the presence of a cyclic alternating pattern and a poorly preserved REM/NREM cyclicity. In relation to total sleep time, percentages of different sleep stages were as follows: 13.4% N1, 47.3% N2, 23.1% N3, and 16.1% REM%. Sleep latency and REM sleep latency were 14.3 minutes and 265 minutes, respectively. There were no significant respiratory events during sleep (apnea-hypopnea index 4.1 events/h) or any periodic movements. Similarly, no epileptiform activity or masturbatory behaviors, or other parasomnia behaviors, were observed during the examination.

The management of the sexsomnia included information about the condition and its consequences, along with management options. Cognitive behavior therapy was initiated, emphasizing the importance of improving sleep hygiene; also, the dose of bedtime melatonin was increased to 3 mg. Any consideration to discontinue azathioprine was contraindicated as the patient experienced a chronic inflammatory bowel disease with prominent symptoms. At clinic follow-up after 3 months, with the patient taking 3 mg of melatonin and fulfilling a regular sleep schedule (8 hours/night), she reported a good clinical response with decreased sleep masturbation episodes per week, and even having several weeks without any such behaviors. The basis for this reported benefit was that the patient continued to sleep with her bed partner who reported the reduction of her sexsomnia behaviors, which she also indirectly confirmed to herself as in the morning she would observe the absence of disarray of her sheets and blanket. However, at a subsequent follow-up appointment, she reported noncompliance with melatonin therapy, as she often did not remember to take melatonin (which did not cause any side effect), and also she eventually failed to comply with the prescribed sleep hygiene measures, coincident with stress related to personal and family issues. These factors led to a worsening of the clinical picture, with increased frequency and intensity of her sexsomnia episodes.

DISCUSSION

This case of sexsomnia is noteworthy for the following reasons.

First, it may be the first reported case of sexsomnia in an adolescent female; one previously reported case of a 17-year-old patient did not specify the sex.⁴ Apart from that case, there are only four other previously reported cases of sexsomnia in an adolescent, all males with the youngest ages 15 and 16 years old.^9,10 One recent series of 65 patients with sexsomnia included a 15-year-old male as the only adolescent case.¹⁰ However, by history, 18 patients reported a childhood onset of sexsomnia, and 3 patients reported an adolescent onset of sexsomnia. A female with sexsomnia onset at age 7 years reported that her parents “found her rubbing her teddy bear against her genitals.” Apart from this isolated description, no other details were provided about sexsomnia behaviors and any psychosocial consequences during childhood and adolescence in this subgroup of 21 preadult-onset patients with sexsomnia. Another recent series of 24 patients with sexsomnia included a 17-year-old, without sex specified, as previously reported in an earlier study by the same group.^4,11 However, among the adult patients, 1 reported a childhood onset of sexsomnia, and 4 patients reported an adolescent onset of sexsomnia. Nevertheless, this series also did not provide any details about the sexsomnia behaviors and any psychosocial consequences during childhood and adolescence in this subgroup of 5 patients with preadult-onset sexsomnia. Therefore, details on psychosocial and physical consequences from sexsomnia in childhood or adolescence, as described in our case, have been not previously been reported.

Second, this case of sexsomnia in a female is an uncommon occurrence, as only 16% of 220 sexsomnia cases in the current published world literature (from 15 countries) are female,¹² with 4 case series and 2 comprehensive reviews comprising a majority of reported cases.^4–7,10,11 Sexsomnia is a male-predominant parasomnia.

Third, the trigger for emergence of sexsomnia was the onset of Crohn’s disease and initiation of azathioprine therapy, which has not previously been reported for sexsomnia (or to our knowledge from any other autoimmune/immune-mediated disease). It is possible, if not probable, that stress associated with the diagnosis and treatment of Crohn’s disease, with important dietary and lifestyle changes, may have also played a role (including a primary role) in triggering the onset of sexsomnia.

Fourth, her initial clinical presentation from the sexsomnia was to a psychiatrist due to depression and “feeling ashamed” from being told by her male partner that she was masturbating during sleep for which she was fully amnestic and for which she was worried about any negative impact on their relationship. This calls attention to the importance of mental health professionals becoming informed about sexsomnia and its negative psychosocial consequences. Embarrassments and worries felt by a patient (as in our case), and/or the intimate partner is common with sexsomnia and has been comprehensively addressed in a recent report on the psychosocial consequences of sexsomnia.¹³

Fifth, well-known predisposing factors for sexsomnia in our case were a prior history of sleepwalking, a NREM parasomnia, along with a positive family history of parasomnia involving the mother with sleepwalking. As described in several studies, sexsomnia may occur along the evolution of other NREM (and occasionally also REM) parasomnias, with sexsomnia being the last parasomnia to appear.⁷ In fact, Dubessy et al showed that 47.6% of the patients had a current or previous history of sleepwalking or night terrors.⁴ However, in our case, why sexsomnia emerged rather than a reactivation of sleepwalking (alone or with de novo sexsomnia) remains unexplained.

Sixth, poor sleep hygiene was a perpetuating factor for the sexsomnia that was controlled with cognitive behavioral therapy and melatonin (after a dose increase). This is a common scenario for NREM parasomnias. Stress-induced relapse of poor sleep hygiene in our patient resulted in relapse of the sexsomnia, another well-known scenario in NREM parasomnias. Thus, the combination of relapsing poor sleep hygiene along with stress could explain the recurrence of her sexsomnia. In fact, there are several cases that show the link between sexsomnia, stress, sleep deprivation, and circadian rhythm disorders.^3–8

Seventh, the patient experienced pain in the pubic area as the first symptom of her sexsomnia, before her male partner witnessed her sleep masturbation while sleeping together. Various injuries (to self and bed partner), at times serious, have been reported with sexsomnia, including vaginal and clitoral injuries in females from prolonged and vigorous masturbation during sleep without awareness, especially when sleeping alone.^3,4,6

The true prevalence of sexsomnia is still unknown.⁵ A Norwegian study conducted in the general population showed a lifetime sexsomnia prevalence of 7.1%.^3,7 As stated earlier, this is a strongly male predominant disorder, and so a case such as ours, involving an adolescent female, affords the opportunity to better understand sexsomnia from a broader clinical perspective. Sexsomnia occurs mainly in young adults but can emerge throughout most of the lifespan.¹²

In conclusion, this challenging case calls attention to the need for sleep clinicians, along with primary care providers and mental health professionals, to become aware that sexsomnia can affect female and male adolescents, with psychosocial and physical consequences that require prompt and effective management.

DISCLOSURE STATEMENT

All authors have seen and approved the manuscript. Work was performed in the Department of Psychiatry and Mental Health, Centro Hospitalar Tondela-Viseu, Viseu, Portugal. Carlos H. Schenck reports a one-time lecture honorarium from Eisai, Inc. The other authors report no conflicts of interest.

ABBREVIATIONS

NREM

non-rapid eye movement

REM

rapid eye movement