C A S E R E P O RT S
pii: jc-00331-14 http://dx.doi.org/10.5664/jcsm.4542
Trichotillomania and Non-Epileptic Seizures as Sleep-Related Dissociative Phenomena
Melina Angulo-Franco, MD; Alejandra Bush-Martínez, MD; Alejandro Nenclares-Portocarrero, MD; Alejandro Jiménez-Genchi, MD, MS Servicios Clínicos, Instituto Nacional de Psiquiatría Ramón de la Fuente Muñiz, Tlalpan, México
The occurrence of non-epileptic seizures (NES) and trichotillomania during sleep is rare. We describe the case of an adult woman with a personal history of childhood maltreatment and psychiatric morbidity (major depression, trichotillomania, and conversion disorder), who was referred to the sleep unit because of nocturnal hair-pulling and psychomotor agitation during sleep. An all-night PSG recording with audiovisual monitoring documented seven episodes of trichotillomania and one NES, all of which arose from unequivocal wakefulness. Improvement
of nocturnal behaviors was observed after long-term psychotherapy. This case illustrates that nocturnal trichotillomania and NES may be symptoms of a sleep-related dissociative disorder. Keywords: trichotillomania, dissociative disorder, parasomnia, convulsion, non-epileptic, sleep Citation: Angulo-Franco M, Bush-Martínez A, NenclaresPortocarrero A, Jiménez-Genchi A. Trichotillomania and non-epileptic seizures as sleep-related dissociative phenomena. J Clin Sleep Med 2015;11(3):271–273.
S
leep-related dissociative disorders (SRDD) are defined as dissociative disorders that can arise throughout the sleep period during well-established wakefulness.1 Nocturnal behavior of patients with SRDD may correspond to the behaviors observed in dissociative disorders (DD; ≥ 2 distinct personality states or a nocturnal fugue). However, some patients have presented nocturnal behaviors that represent reenactments of past traumatic incidents, self-mutilating behaviors, violent behavior, and psychogenic events.1,2 This suggests that behaviors in SRDD may resemble daytime DD but are not limited to them. Herein we describe the case of a patient with sleep-related trichotillomania and psychogenic non-epileptic seizures (NES) as symptoms of nocturnal dissociation.
psychotherapy produced significant improvement. After two years, she was referred to the sleep clinic because on waking up in the morning she discovered she was holding hairs in her hands and there were also many hairs over the bed. According to her daughter, Mrs. B would often show hairpulling during the night but had no recollection whatsoever of this behavior; however, sometimes she would dream about the hair-pulling act. She also complained of insomnia, nightmare-related psychomotor agitation, and exhibited violent behavior. A polysomnographic recording with audiovisual monitoring was performed. Ninety minutes after sleep onset, she had an arousal, and 90 sec later the patient initiated a distinctive hairpulling behavior (Figure 1 and Video 1). First she selected one hair and pulled it out from her scalp; she then put the hair in front of her face and tried to straighten it up; afterwards, she placed it inside her left ear for a few seconds before finally throwing it away. She had 7 hair-pulling events of similar characteristics throughout the night. All of the events arose from an EEG wakefulness state with alpha rhythm preceding the episodes; no epileptiform activity was noted. These awakenings were preceded by different NREM sleep stages (Figure 2), and they were not associated with respiratory events or leg movements (both the apnea-hypopnea index of 1.6/h and the periodic limb movements of sleep index 1.3/h were normal). Six hours 50 minutes after sleep onset she presented an episode characterized by flailing movements, beginning with the legs, then the arms, and finally the whole body; the episode finished with head-banging movements. Moaning and cries were present during the entire 143-sec attack. This episode arose from a wakefulness state, and no epileptic discharges were observed. During the follow-up, NES and hair-pulling showed very slow improvement until remission after two years in psychotherapy and attendance to a self-help group.
REPORT OF CASE Mrs. B is a 41-year-old woman with a personal history of severe childhood maltreatment. In association to childhood abuse, she experienced episodes of intense anxiety, which sometimes triggered seizure-like events characterized by tremor-like movements of the jaw, then in the arms and legs, and finally in the whole body. After these events, she frequently presented a distinctive hair-pulling behavior. First, she pulled hair out from her scalp one by one until she had approximately 40 in her hand. Then she felt the urge to introduce them into the ear; after this, she manipulated them trying to separate them into two halves. The ritual ended when she swallowed hair or threw it away. When she was 39 years old, she came to our institution because of persistent anxiety and depressive symptoms and frequent seizure-like events. A major depressive disorder, conversion disorder and trichotillomania were identified. A routine EEG and a brain CT scan showed no abnormalities. Treatment with sertraline, clonazepam, and supportive 271
Journal of Clinical Sleep Medicine, Vol. 11, No. 3, 2015
M Angulo-Franco, A Bush-Martínez, A Nenclares-Portocarrero et al.
Figure 1—The photographic sequence shows a ritual of sleep-related trichotillomania.
First, the patient selects one hair (upper left); then she straightens it out (upper right) and places it inside her left ear (bottom left); finally she throws it away (bottom right). The PSG of the first minute of the hair-pulling episode shows artifacts corresponding to the moments when the patient touches her head to select one hair, superimposed on a background of alpha rhythm.
12 patients who were apparently sleeping.3 More recently, it has been found that up to 13% of NES episodes occur during apparent sleep.4 In the case we report, several features suggestive of NES are present, such as the occurrence during pseudo-sleep, flailing and head movements, the presence of moaning and crying during the seizure, and long duration.4 Although DSM-5 classifies NES as a conversion disorder, the
DISCUSSION Although trichotillomania and NES have been previously reported as behaviors which might arise during sleep, the cooccurrence of both has not been described. In an early study, Thacker et al. documented with video-EEG the occurrence of NES during well-established wakefulness in Journal of Clinical Sleep Medicine, Vol. 11, No. 3, 2015
272
Case Report
Figure 2
Note that both trichotillomania (blue blocks) and non-epileptic seizures (green block) events occurred during wakefulness. Four hair-pulling rituals occurred during awakenings preceded by different NREM sleep stages and the rest after long waking states.
close relationship between conversion and dissociation is well-known. Regarding hair-pulling, results from a survey among dermatologists suggest that sleep-isolated trichotillomania is not uncommon.5 In fact, Murphy et al. reported the case of a woman with hair-pulling occurring exclusively in NREM sleep.6 In the case we are describing, trichotillomania occurred as a dissociative phenomenon. Although the patient presented with two hair-pulling episodes a few minutes after the end of stages N3 sleep, all the behaviors arose during unequivocal wakefulness, and 3 episodes were preceded by long periods (> 15 min) of a complete waking state. In addition to this, the patient presented several characteristics suggestive of SRDD, such as being female, a history of childhood abuse, psychiatric morbidity, and daytime dissociative symptoms.1 Even though SRDD is no longer considered as a parasomnia in the third edition of the International Classification of Sleep Disorders, it is still included in the differential diagnosis of disorders of arousal and REM sleep behavior disorder. Factors responsible for SRDD are unknown. However, recent research has found a strong association between trait dissociation, psychological distress, and unusual sleep and dream experiences.7
3. Thacker K, Devinsky O, Perrine K, Alper K, Luciano D. Nonepileptic seizures during apparent sleep. Ann Neurol 1993;33:414–8. 4. Devinsky O, Gazzola D, LaFrance WC Jr. Differentiating between nonepileptic and epileptic seizures. Nat Rev Neurol 2011;7:210–20. 5. Murphy C, Redenius R, O’Neill E, et al. Sleep-isolated trichotillomania: a survey of dermatologists. J Clin Sleep Med 2007;3:719–21. 6. Murphy C, Valerio T, Zallek S. Trichotillomania: a NREM sleep parasomnia? Neurology 2006;66:1276. 7. Soffer-Dudek N, Shahar G. What are sleep-related experiences? Associations with transliminality, psychological distress, and life stress. Conscious Cogn 2009;18:891–904.
ACKNOWLEDGMENTS The authors thank Luis Rodríguez-Avilés and Fernando Jiménez-Peña for their assistance with the elaboration of the figures and video edition.
SUBMISSION & CORRESPONDENCE INFORMATION Submitted for publication August, 2014 Submitted in final revised form November, 2014 Accepted for publication November, 2014 Address correspondence to: Alejandro Jiménez-Genchi, Instituto Nacional de Psiquiatría Ramón de la Fuente Muñiz, Servicios Clínicos, Calz. México-Xochimilco 101, Col. San Lorenzo Huipulco C. P. 14370, Tlalpan México, D. F.; Tel: 41 60 53 48; Email: [email protected], [email protected]
DISCLOSURE STATEMENT
REFERENCES
This work was performed at the Instituto Nacional de Psiquiatría Ramón de la Fuente Muñiz and it was made without financial support. Dr. Jiménez-Genchi has served on advisory boards and as a speaker for Sanofi Aventis Mexico and has developed educational material for Roche México. The other authors have indicated no financial conflicts of interest.
1. American Academy of Sleep Medicine. The international classification of sleep disorders: diagnostic and coding manual. 2nd ed. Westchester, IL: American Academy of Sleep Medicine, 2005. 2. Agargun MY, Kara H, Özer OA, et al. Characteristics of patients with nocturnal dissociative disorders. Sleep Hypn 2001;3:131–4.
273
Journal of Clinical Sleep Medicine, Vol. 11, No. 3, 2015
pii: jc-00331-14 http://dx.doi.org/10.5664/jcsm.4542
Trichotillomania and Non-Epileptic Seizures as Sleep-Related Dissociative Phenomena
Melina Angulo-Franco, MD; Alejandra Bush-Martínez, MD; Alejandro Nenclares-Portocarrero, MD; Alejandro Jiménez-Genchi, MD, MS Servicios Clínicos, Instituto Nacional de Psiquiatría Ramón de la Fuente Muñiz, Tlalpan, México
The occurrence of non-epileptic seizures (NES) and trichotillomania during sleep is rare. We describe the case of an adult woman with a personal history of childhood maltreatment and psychiatric morbidity (major depression, trichotillomania, and conversion disorder), who was referred to the sleep unit because of nocturnal hair-pulling and psychomotor agitation during sleep. An all-night PSG recording with audiovisual monitoring documented seven episodes of trichotillomania and one NES, all of which arose from unequivocal wakefulness. Improvement
of nocturnal behaviors was observed after long-term psychotherapy. This case illustrates that nocturnal trichotillomania and NES may be symptoms of a sleep-related dissociative disorder. Keywords: trichotillomania, dissociative disorder, parasomnia, convulsion, non-epileptic, sleep Citation: Angulo-Franco M, Bush-Martínez A, NenclaresPortocarrero A, Jiménez-Genchi A. Trichotillomania and non-epileptic seizures as sleep-related dissociative phenomena. J Clin Sleep Med 2015;11(3):271–273.
S
leep-related dissociative disorders (SRDD) are defined as dissociative disorders that can arise throughout the sleep period during well-established wakefulness.1 Nocturnal behavior of patients with SRDD may correspond to the behaviors observed in dissociative disorders (DD; ≥ 2 distinct personality states or a nocturnal fugue). However, some patients have presented nocturnal behaviors that represent reenactments of past traumatic incidents, self-mutilating behaviors, violent behavior, and psychogenic events.1,2 This suggests that behaviors in SRDD may resemble daytime DD but are not limited to them. Herein we describe the case of a patient with sleep-related trichotillomania and psychogenic non-epileptic seizures (NES) as symptoms of nocturnal dissociation.
psychotherapy produced significant improvement. After two years, she was referred to the sleep clinic because on waking up in the morning she discovered she was holding hairs in her hands and there were also many hairs over the bed. According to her daughter, Mrs. B would often show hairpulling during the night but had no recollection whatsoever of this behavior; however, sometimes she would dream about the hair-pulling act. She also complained of insomnia, nightmare-related psychomotor agitation, and exhibited violent behavior. A polysomnographic recording with audiovisual monitoring was performed. Ninety minutes after sleep onset, she had an arousal, and 90 sec later the patient initiated a distinctive hairpulling behavior (Figure 1 and Video 1). First she selected one hair and pulled it out from her scalp; she then put the hair in front of her face and tried to straighten it up; afterwards, she placed it inside her left ear for a few seconds before finally throwing it away. She had 7 hair-pulling events of similar characteristics throughout the night. All of the events arose from an EEG wakefulness state with alpha rhythm preceding the episodes; no epileptiform activity was noted. These awakenings were preceded by different NREM sleep stages (Figure 2), and they were not associated with respiratory events or leg movements (both the apnea-hypopnea index of 1.6/h and the periodic limb movements of sleep index 1.3/h were normal). Six hours 50 minutes after sleep onset she presented an episode characterized by flailing movements, beginning with the legs, then the arms, and finally the whole body; the episode finished with head-banging movements. Moaning and cries were present during the entire 143-sec attack. This episode arose from a wakefulness state, and no epileptic discharges were observed. During the follow-up, NES and hair-pulling showed very slow improvement until remission after two years in psychotherapy and attendance to a self-help group.
REPORT OF CASE Mrs. B is a 41-year-old woman with a personal history of severe childhood maltreatment. In association to childhood abuse, she experienced episodes of intense anxiety, which sometimes triggered seizure-like events characterized by tremor-like movements of the jaw, then in the arms and legs, and finally in the whole body. After these events, she frequently presented a distinctive hair-pulling behavior. First, she pulled hair out from her scalp one by one until she had approximately 40 in her hand. Then she felt the urge to introduce them into the ear; after this, she manipulated them trying to separate them into two halves. The ritual ended when she swallowed hair or threw it away. When she was 39 years old, she came to our institution because of persistent anxiety and depressive symptoms and frequent seizure-like events. A major depressive disorder, conversion disorder and trichotillomania were identified. A routine EEG and a brain CT scan showed no abnormalities. Treatment with sertraline, clonazepam, and supportive 271
Journal of Clinical Sleep Medicine, Vol. 11, No. 3, 2015
M Angulo-Franco, A Bush-Martínez, A Nenclares-Portocarrero et al.
Figure 1—The photographic sequence shows a ritual of sleep-related trichotillomania.
First, the patient selects one hair (upper left); then she straightens it out (upper right) and places it inside her left ear (bottom left); finally she throws it away (bottom right). The PSG of the first minute of the hair-pulling episode shows artifacts corresponding to the moments when the patient touches her head to select one hair, superimposed on a background of alpha rhythm.
12 patients who were apparently sleeping.3 More recently, it has been found that up to 13% of NES episodes occur during apparent sleep.4 In the case we report, several features suggestive of NES are present, such as the occurrence during pseudo-sleep, flailing and head movements, the presence of moaning and crying during the seizure, and long duration.4 Although DSM-5 classifies NES as a conversion disorder, the
DISCUSSION Although trichotillomania and NES have been previously reported as behaviors which might arise during sleep, the cooccurrence of both has not been described. In an early study, Thacker et al. documented with video-EEG the occurrence of NES during well-established wakefulness in Journal of Clinical Sleep Medicine, Vol. 11, No. 3, 2015
272
Case Report
Figure 2
Note that both trichotillomania (blue blocks) and non-epileptic seizures (green block) events occurred during wakefulness. Four hair-pulling rituals occurred during awakenings preceded by different NREM sleep stages and the rest after long waking states.
close relationship between conversion and dissociation is well-known. Regarding hair-pulling, results from a survey among dermatologists suggest that sleep-isolated trichotillomania is not uncommon.5 In fact, Murphy et al. reported the case of a woman with hair-pulling occurring exclusively in NREM sleep.6 In the case we are describing, trichotillomania occurred as a dissociative phenomenon. Although the patient presented with two hair-pulling episodes a few minutes after the end of stages N3 sleep, all the behaviors arose during unequivocal wakefulness, and 3 episodes were preceded by long periods (> 15 min) of a complete waking state. In addition to this, the patient presented several characteristics suggestive of SRDD, such as being female, a history of childhood abuse, psychiatric morbidity, and daytime dissociative symptoms.1 Even though SRDD is no longer considered as a parasomnia in the third edition of the International Classification of Sleep Disorders, it is still included in the differential diagnosis of disorders of arousal and REM sleep behavior disorder. Factors responsible for SRDD are unknown. However, recent research has found a strong association between trait dissociation, psychological distress, and unusual sleep and dream experiences.7
3. Thacker K, Devinsky O, Perrine K, Alper K, Luciano D. Nonepileptic seizures during apparent sleep. Ann Neurol 1993;33:414–8. 4. Devinsky O, Gazzola D, LaFrance WC Jr. Differentiating between nonepileptic and epileptic seizures. Nat Rev Neurol 2011;7:210–20. 5. Murphy C, Redenius R, O’Neill E, et al. Sleep-isolated trichotillomania: a survey of dermatologists. J Clin Sleep Med 2007;3:719–21. 6. Murphy C, Valerio T, Zallek S. Trichotillomania: a NREM sleep parasomnia? Neurology 2006;66:1276. 7. Soffer-Dudek N, Shahar G. What are sleep-related experiences? Associations with transliminality, psychological distress, and life stress. Conscious Cogn 2009;18:891–904.
ACKNOWLEDGMENTS The authors thank Luis Rodríguez-Avilés and Fernando Jiménez-Peña for their assistance with the elaboration of the figures and video edition.
SUBMISSION & CORRESPONDENCE INFORMATION Submitted for publication August, 2014 Submitted in final revised form November, 2014 Accepted for publication November, 2014 Address correspondence to: Alejandro Jiménez-Genchi, Instituto Nacional de Psiquiatría Ramón de la Fuente Muñiz, Servicios Clínicos, Calz. México-Xochimilco 101, Col. San Lorenzo Huipulco C. P. 14370, Tlalpan México, D. F.; Tel: 41 60 53 48; Email: [email protected], [email protected]
DISCLOSURE STATEMENT
REFERENCES
This work was performed at the Instituto Nacional de Psiquiatría Ramón de la Fuente Muñiz and it was made without financial support. Dr. Jiménez-Genchi has served on advisory boards and as a speaker for Sanofi Aventis Mexico and has developed educational material for Roche México. The other authors have indicated no financial conflicts of interest.
1. American Academy of Sleep Medicine. The international classification of sleep disorders: diagnostic and coding manual. 2nd ed. Westchester, IL: American Academy of Sleep Medicine, 2005. 2. Agargun MY, Kara H, Özer OA, et al. Characteristics of patients with nocturnal dissociative disorders. Sleep Hypn 2001;3:131–4.
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Journal of Clinical Sleep Medicine, Vol. 11, No. 3, 2015
