Nonconvulsive status epilepticus manifesting as full-blown psychosis during pregnancy
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Clinical letter Nonconvulsive status epilepticus manifesting as full-blown psychosis during pregnancy Doh-Eui Kim 1 , Yang-Je Cho, Moon Kyu Lee 2 , Byung In Lee, Kyoung Heo * Department of Neurology, Epilepsy Research Institute, Yonsei University College of Medicine, 50 Yonsei-ro, Seodaemun-gu, Seoul 120-752, Republic of Korea 1. Introduction Nonconvulsive status epilepticus (NCSE) can produce a wide spectrum of clinical symptoms depending on the regions and the relative amount of brain that is made dysfunctional by persistent epileptic discharges. 1 NCSE can cause ictal behaviors resembling psychosis. However, only a few clear cases of NCSE causing diverse features of psychosis have been reported. 1 We describe a patient with NCSE during pregnancy, who demonstrated full-blown psychotic symptoms associated with right hemispheric ictal activity on EEG. 2. Case report A 34-year-old, right-handed woman suffered from viral encephalitis with seizures at 22 years of age. She made a good cognitive recovery and was able to return to a normal social life. The patient chose to stop taking antiepileptic drugs (AEDs). Once or twice a month the patient experienced brief convulsive seizures during sleep involving the left side of her face. She did not experience any psychiatric problems requiring treatment at this time. Twelve years later, the patient was admitted with six to seven episodes of left facial twitching per day and after experiencing a single generalized tonic-clonic seizure. She was found to be 6 weeks pregnant. MRI did not show any abnormalities. The EEG revealed low-voltage arrhythmic delta waves (LVAD) in the right hemisphere. The left facial twitching was triggered by articulation and mastication, and was not found to be associated with any visually identifiable ictal scalp EEG changes. Nevertheless the episodes of facial twitching were interpreted as focal epileptic seizures. These seizures were well controlled by carbamazepine at 600 mg/day for three days, after which the patient was discharged. She became anxious and agitated within hours of discharge, and developed various psychotic symptoms. She was re-admitted eight days after discharge for video-EEG monitoring. She reported an intermittent, transient homonymous visual field defect affecting the left lower quadrants, the illusion of visual distortion on the left side, visual hallucinations of ghosts, out-of-body experiences, and panic attacks with choking sensations. She exhibited unusual mannerisms such as staring vacantly and moving her left arm up and down repetitively, apparently compulsive behaviors that included repeated brushing and rinsing of the tongue, and monotonous speech. She stated that the voice of her mother had changed and sounded like a robot. She also complained that her mother, along with the doctors, was blaming her and trying to control her. She further complained that the nurses were trying to give her harmful pills and take her baby away from her. Sometimes, she was not able to control her anger and quarreled with her parents. While exhibiting these psychotic symptoms, she was fully oriented, correctly performed a serial- seven test, and had no left-right disorientation. Her language functions including naming, writing, speaking, and repetition were intact. During sleep, she was observed to have left facial twitching. Continuous video-EEG monitoring showed prolonged episodes of high-voltage 1.5–2 Hz rhythmic delta activities (HVRD) in the right hemisphere that persisted for one to three hours (Fig. 1A), and changed to LVAD for brief periods. While EEG revealed LVAD (Fig. 1B), the patient’s psychotic symptoms showed temporary improvement. Additionally, the patient remembered most of the events that occurred during the period characterized by HVRD, and she apologized to her parents and doctors about her previous behavior. Seventeen hours after the beginning of video-EEG monitoring, she was treated with an intravenous midazolam infusion along with carbamazepine, pregabalin, and topiramate. After a gradual escalation to 0.3 mg/kg/h of midazolam, HVRD were controlled for 2 days, at which point the midazolam infusion was slowly tapered Seizure 23 (2014) 402–404 A R T I C L E I N F O Article history: Received 10 August 2013 Received in revised form 14 January 2014 Accepted 16 January 2014 * Corresponding author. Tel.: +82 2 2228 1607; fax: +82 2 393 0705. E-mail address: [email protected] (K. Heo). 1 Present address: Department of Neurology, Sunchunhyang University College of Medicine, Cheonan Hospital, 31 Soonchunhyang 6 gil, Dongnam-gu, Cheonan 330-721, Republic of Korea. 2 Present address: Department of Neurology, University of Ulsan College of Medicine, Gangeung Asan Hospital, 38 Bangdong-gil, Sacheon-myeon, Gangneung 210-711, Republic of Korea. Contents lists available at ScienceDirect Seizure jou r nal h o mep age: w ww.els evier .co m/lo c ate/ys eiz 1059-1311/$ – see front matter ß 2014 British Epilepsy Association. Published by Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.seizure.2014.01.020
Transcript of Nonconvulsive status epilepticus manifesting as full-blown psychosis during pregnancy
Seizure 23 (2014) 402–404
Clinical letter
Nonconvulsive status epilepticus manifesting as full-blown psychosisduring pregnancy
Doh-Eui Kim 1, Yang-Je Cho, Moon Kyu Lee 2, Byung In Lee, Kyoung Heo *
Department of Neurology, Epilepsy Research Institute, Yonsei University College of Medicine, 50 Yonsei-ro, Seodaemun-gu, Seoul 120-752, Republic of Korea
Contents lists available at ScienceDirect
Seizure
jou r nal h o mep age: w ww.els evier . co m/lo c ate /ys eiz
A R T I C L E I N F O
Article history:
Received 10 August 2013
Received in revised form 14 January 2014
Accepted 16 January 2014
1. Introduction
Nonconvulsive status epilepticus (NCSE) can produce a widespectrum of clinical symptoms depending on the regions and therelative amount of brain that is made dysfunctional by persistentepileptic discharges.1 NCSE can cause ictal behaviors resemblingpsychosis. However, only a few clear cases of NCSE causing diversefeatures of psychosis have been reported.1 We describe a patientwith NCSE during pregnancy, who demonstrated full-blownpsychotic symptoms associated with right hemispheric ictalactivity on EEG.
2. Case report
A 34-year-old, right-handed woman suffered from viralencephalitis with seizures at 22 years of age. She made a goodcognitive recovery and was able to return to a normal social life.The patient chose to stop taking antiepileptic drugs (AEDs). Once ortwice a month the patient experienced brief convulsive seizuresduring sleep involving the left side of her face. She did notexperience any psychiatric problems requiring treatment at thistime.
Twelve years later, the patient was admitted with six to sevenepisodes of left facial twitching per day and after experiencinga single generalized tonic-clonic seizure. She was found to be6 weeks pregnant. MRI did not show any abnormalities. The EEG
* Corresponding author. Tel.: +82 2 2228 1607; fax: +82 2 393 0705.
E-mail address: [email protected] (K. Heo).1 Present address: Department of Neurology, Sunchunhyang University College
of Medicine, Cheonan Hospital, 31 Soonchunhyang 6 gil, Dongnam-gu, Cheonan
330-721, Republic of Korea.2 Present address: Department of Neurology, University of Ulsan College of
Medicine, Gangeung Asan Hospital, 38 Bangdong-gil, Sacheon-myeon, Gangneung
210-711, Republic of Korea.
1059-1311/$ – see front matter � 2014 British Epilepsy Association. Published by Else
http://dx.doi.org/10.1016/j.seizure.2014.01.020
revealed low-voltage arrhythmic delta waves (LVAD) in the righthemisphere. The left facial twitching was triggered by articulationand mastication, and was not found to be associated with anyvisually identifiable ictal scalp EEG changes. Nevertheless theepisodes of facial twitching were interpreted as focal epilepticseizures. These seizures were well controlled by carbamazepineat 600 mg/day for three days, after which the patient wasdischarged.
She became anxious and agitated within hours of discharge,and developed various psychotic symptoms. She was re-admittedeight days after discharge for video-EEG monitoring. She reportedan intermittent, transient homonymous visual field defectaffecting the left lower quadrants, the illusion of visual distortionon the left side, visual hallucinations of ghosts, out-of-bodyexperiences, and panic attacks with choking sensations. Sheexhibited unusual mannerisms such as staring vacantly andmoving her left arm up and down repetitively, apparentlycompulsive behaviors that included repeated brushing andrinsing of the tongue, and monotonous speech. She stated thatthe voice of her mother had changed and sounded like a robot. Shealso complained that her mother, along with the doctors, wasblaming her and trying to control her. She further complained thatthe nurses were trying to give her harmful pills and take her babyaway from her. Sometimes, she was not able to control her angerand quarreled with her parents. While exhibiting these psychoticsymptoms, she was fully oriented, correctly performed a serial-seven test, and had no left-right disorientation. Her languagefunctions including naming, writing, speaking, and repetitionwere intact. During sleep, she was observed to have left facialtwitching.
Continuous video-EEG monitoring showed prolonged episodesof high-voltage 1.5–2 Hz rhythmic delta activities (HVRD) in theright hemisphere that persisted for one to three hours (Fig. 1A), andchanged to LVAD for brief periods. While EEG revealed LVAD(Fig. 1B), the patient’s psychotic symptoms showed temporaryimprovement. Additionally, the patient remembered most of theevents that occurred during the period characterized by HVRD, andshe apologized to her parents and doctors about her previousbehavior.
Seventeen hours after the beginning of video-EEG monitoring,she was treated with an intravenous midazolam infusion alongwith carbamazepine, pregabalin, and topiramate. After a gradualescalation to 0.3 mg/kg/h of midazolam, HVRD were controlled for2 days, at which point the midazolam infusion was slowly tapered
vier Ltd. All rights reserved.
Fig. 1. (A) During psychotic symptoms, an EEG revealed high-voltage 1.5–2 Hz rhythmic delta activities in the right hemisphere. (B) During temporary improvement of
psychotic symptoms, EEG showed low voltage arrhythmic delta waves in the right hemisphere.
Fig. 2. 99mTc-HMPAO SPECT. (A) Ictal SPECT demonstrates hyperperfusion in almost the entire area of the right hemisphere. (B) Interictal SPECT demonstrates hypoperfusion
in the right hemisphere.
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out. However, psychotic symptoms and HVRD reappeared.Meanwhile, a subchorionic hematoma was detected in this patientand deep vein thrombosis developed in the left common iliac vein,requiring anticoagulation therapy and insertion of an inferior venacava filter. On the 10th day of admission, the pregnancy wasterminated. Her psychotic symptoms and HVRD, however, did notimmediately subside, and she underwent 99mTc-hexamethylpro-pyleneamine oxime single-photon emission computed tomogra-phy (99mTc-HMPAO SPECT). This test showed hyperperfusionaffecting almost the entire right hemisphere (Fig. 2A). Themidazolam infusion was restarted. The frequency and durationof her psychotic symptoms gradually improved after the mid-azolam treatment, which was tapered over the course of six days.An EEG taken prior to discharge when she did not show psychoticsymptoms showed only LVAD. The patient’s psychotic symptomsdisappeared over the next 2 months, although she was stillreported as having left facial twitching during sleep 3–4 times permonth. Nine months later, interictal SPECT demonstrated hypo-perfusion affecting almost the entire right hemisphere (Fig. 2B).
3. Discussion
Herein, we report the case of a patient who experienced aprolonged period of variable psychotic symptoms associated withHVRD. HVRD did not exhibit the evolving pattern more typically
seen in an ictal EEG. Nevertheless, the changes seen wereconsidered highly suggestive of an ictal EEG pattern of NCSE,especially considering the temporal association with the patient’sclinical symptoms, her clinical and EEG response to midazolamtreatment and the right hemispheric hyperperfusion on SPECT.
Previous reports of ictal psychotic symptoms have demon-strated that the nature of a patient’s particular psychoticsymptoms depends on the anatomical areas of ictal origin orinvolvement, particularly in the language nondominant hemi-sphere. In one previous case, the occurrence of paranoid psychoticsymptom was associated with frequent sharp waves in the righttemporal region.2 Another previous report described panic attack-like fear due to ictal discharges that arose from the rightparietotemporal region related to a surgical scar.3 Out-of-bodysensations have been reported in seizures arising from thenondominant parietal region which plays a role in normalintegration of body representation.4
Our patient presented with full-blown psychotic symptoms.Consciousness (alertness, attention, orientation, and memory) wasmaintained throughout her psychotic symptoms. NCSE likelyoriginated from the right motor area, and although we were unableto exactly delineate the extent of ictal involvement, based on theSPECT findings, it possibly involved almost the entire righthemisphere, which may explain the diverse nature of her psychoticsymptoms. Our patient had had a relatively stable epilepsy not
D.-E. Kim et al. / Seizure 23 (2014) 402–404404
requiring AED treatment for a long period of time prior to the onsetof her ictal psychotic episode. Pregnancy-related hormonalchanges may be one plausible explanation for our patient’s seizureexacerbation.
Conflict of interest
None.
References
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2. Trimble MR. The psychoses of epilepsy. New York: Raven Press; 1991.3. Brigo F, Ferlisi M, Fiaschi A, Bongiovanni LG. Fear as the only clinical expression of
affective focal status epilepticus. Epilepsy & Behavior 2011;20:107–10.4. Maillard L, Vignal JP, Anxionnat R, Taillandier L, Vespignani H. Semiologic value
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