Video-Clinical Corners Rhythmic movement disorder associated with respiratory arousals and improved by CPAP titration in a patient with restless legs syndrome and sleep apnea Parham Gharagozlou * , Michael Seyffert, Roberto Santos, Sudhansu Chokroverty New Jersey Neuroscience Institute, Seton Hall University, 65 James Street, Edison, NJ 08818, USA Received 30 December 2008; received in revised form 31 January 2009; accepted 9 March 2009 Available online 11 April 2009 Keywords: Rhythmic Movement Disorder; Video-polysomnogram; Sleep apnea; Restless legs syndrome; CPAP titration; Respiratory arousals 1. Introduction Rhythmic Movement Disorder (RMD) is a movement disorder of sleep that is characterized by rhythmic oscilla- tions of the head or limbs [1]. Typically, these stereotyped movements manifest as head banging, body-rocking or head-rolling during sleep. The etiology of RMD is unknown but is hypothesized to be a phenomenon of dis- ordered wake-sleep state transitions [2]. While RMD is usually seen in childhood it is not an uncommon condi- tion in adults [3–6]. It has often been described in both healthy individuals and individuals with neurodevelop- mental or neuropsychiatric disorders, but has rarely been observed in the context of sleep disordered breathing. Here we describe a case of RMD in an adult with video-polysomnographic (video-PSG) documentation, associated with a combination of apneic/hypopneic respiratory events and arousals that are linked to these movements. Following successful continuous positive airway pressure (CPAP) titration, these rhythmic move- ments resolved. 2. The case A 51-year-old man with a past medical history of hypertension, diabetes and hypercholesterolemia pre- sented with snoring, excessive daytime sleepiness and excessive body movements during sleep. He also had a long-standing history of Restless Legs Syndrome (RLS), with ‘‘augmentation” of his symptoms for 1– 2 years prior to the visit. He did not find his sleep refreshing, despite getting about 7 h of sleep per night, and he felt tired throughout the day with an Epworth sleepiness score of 14. After switching RLS medications and spacing out the doses in the evening, his daytime sleepiness improved. His other complaint included head-rolling movements during sleep since childhood. A physical examination revealed an obese man with a BMI of 35 kg/m 2 and a crowded upper airway. The rest of the examination was unremarkable. A split-night PSG-CPAP titration study with recordings from multi- ple cranial and limb muscles was performed. In the pre-CPAP part of the study the patient slept 89 min with a sleep efficiency of 78% and wake time after sleep onset (WASO) of 28 min. His sleep onset latency was 9.5 min. He had an apnea/hypopnea index (AHI) of 91.6 per hour with an arousal index of 89 per hour. No rapid eye movement (REM) sleep or slow wave sleep was noted during the recording. Video recording (see online video segment) and PSG documented rhythmic move- ments (body and head-rolling) that are generally associ- ated with arousals following termination of an apneic or hypopneic event. We observed 13 episodes of rhythmic movements. The movements were primarily body rolling with flexion-extension of the left wrist. Each episode lasted for 4–19 s and was mainly observed while the patient was sleeping on the right side. Frequent head- 1389-9457/$ - see front matter Ó 2009 Elsevier B.V. All rights reserved. doi:10.1016/j.sleep.2009.03.003 * Corresponding author. E-mail address: parhamgr@hotmail.com (P. Gharagozlou). www.elsevier.com/locate/sleep Sleep Medicine 10 (2009) 501–503