Case report An unusual case of episodic stupor Deidre Anne De Silva a, * , Lim Li Ling b , Ng Beng Yeong c , K Puvanendran b a Department of Neurology, National Neuroscience Institute, Singapore General Hospital Campus, Outram Road, Singapore, Singapore 169608 b Department of Neurology, National Neuroscience Institute and Sleep Disorders Unit, Singapore General Hospital Campus, Singapore General Hospital, Outram Road, Singapore, Singapore 169608 c Department of Behavioural Medicine and Sleep Disorders Unit, Singapore General Hospital, Outram Road, Singapore, Singapore 169608 Received 3 November 2005; received in revised form 20 February 2006; accepted 22 February 2006 Abstract We present a case of episodic stupor associated with a myriad of neuropyschiatric manifestations that baffled doctors until they were recognized as sleep attacks. Continuous monitoring and recognition of a cyclical pattern of symptoms and signs helped to uncover the underlying cause of a rapidly cycling bipolar disorder. The symptoms abated quickly and persistently when treated by olanzapine and lithium. q 2006 Elsevier B.V. All rights reserved. Keywords: Sleep attacks; Sleep deprivation; Bipolar disorder; Depression; Hypomania; Stupor 1. Introduction Sleep attacks are an under-recognized cause of episodic stupor. This case highlights that the failure to consider sleep attacks can result to a delay in diagnosis. A detailed history, continuous clinical and polysomnography monitoring led to recognition of the clinical presentation as sleep attacks as well as diagnosis of a bipolar disorder as the underlying cause. 2. Case report Mr J, a 47-year-old Sri Lankan engineer presented with episodic stupor over several months. During one of these episodes, he rammed his car four times in a day and was admitted to hospital in an obtunded state. He also suffered from transient slurring of speech, unsteady gait, double vision and facial droop. He had no significant past medical history and was not on any medications. Two of his cousins suffer from parkinsonism. Mr J is married with two children, does not smoke and infrequently drinks alcohol. The referring doctor did not find any focal neurological deficits. Routine laboratory studies, brain computed tomography (CT) and magnetic resonance imaging (MRI) and inter-event electroencephalogram (EEG) were normal. The symptoms were attributed to vertebrobasilar insuffi- ciency and antiplatelet therapy was started. The episodes of stupor continued and were now associated with Salaam-like attacks. The diagnosis was changed to epilepsy and antiepileptic treatment (sodium valproate) was given. Three months after initial presentation, a second opinion was sought in Singapore, as the symptoms were unabating. Mr J’s son described episodes of unresponsiveness from which arousal was possible but difficult. Our first impression was that these episodes of stupor might be sleep attacks. On initial examination, Mr J was alert and oriented. Cardiovascular, respiratory and abdominal examinations were normal. Tone, power, reflexes and sensation were normal. There were no cranial nerve deficits, nystagmus, cerebellar deficits or cortical signs. Hemogram, renal, liver and thyroid function, repeat brain CT and MRI/MRA and carotid ultrasonography were normal. Toxicological testing was not performed. Nocturnal video polysomnography (PSG) on the first night revealed total sleep time of 7 h 41 min, with 74% sleep efficiency. Sleep latency was 8 min. Rapid eye movement (REM) sleep latency was 63 min. There was 8% slow wave sleep and 27% REM sleep. The apnea-hypopnea index Sleep Medicine 7 (2006) 380–381 www.elsevier.com/locate/sleep 1389-9457/$ - see front matter q 2006 Elsevier B.V. All rights reserved. doi:10.1016/j.sleep.2006.02.008 * Corresponding author. Tel.: C65 63265003; fax: C65 62203321. E-mail address: gnrdsd@sgh.com.sg (D.A. De Silva).