Sleep Medicine
Volume 10, Issue 4 , Pages 501-503, April 2009

Rhythmic movement disorder associated with respiratory arousals and improved by CPAP titration in a patient with restless legs syndrome and sleep apnea

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.

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Keywords: Rhythmic Movement Disorder, Video-polysomnogram, Sleep apnea, Restless legs syndrome, CPAP titration, Respiratory arousals

 

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1. Introduction 

Rhythmic Movement Disorder (RMD) is a movement disorder of sleep that is characterized by rhythmic oscillations 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 disordered wake-sleep state transitions [2]. While RMD is usually seen in childhood it is not an uncommon condition in adults [3], [4], [5], [6]. It has often been described in both healthy individuals and individuals with neurodevelopmental 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 movements resolved.

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2. The case 

A 51-year-old man with a past medical history of hypertension, diabetes and hypercholesterolemia presented 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 1–2years prior to the visit. He did not find his sleep refreshing, despite getting about 7h 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 35kg/m2 and a crowded upper airway. The rest of the examination was unremarkable. A split-night PSG-CPAP titration study with recordings from multiple cranial and limb muscles was performed. In the pre-CPAP part of the study the patient slept 89min with a sleep efficiency of 78% and wake time after sleep onset (WASO) of 28min. His sleep onset latency was 9.5min. 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 movements (body and head-rolling) that are generally associated 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 4–19s and was mainly observed while the patient was sleeping on the right side. Frequent head-rolling movements were also observed. Electromyographic (EMG) recordings showed that most of the rhythmic movements started in right biceps and triceps and propagated to other muscles (Fig. 1, Fig. 2). The majority of EMG bursts lasted between 250 and 450ms. The rhythmic movements varied between 0.5 and 2Hz as part of a cluster of more than four movements. In addition, the patient also had periodic limb movements in sleep (PLMS) and aperiodic EMG bursts. In the CPAP portion of the study the patient had one cluster of periodic and a few aperiodic movements triggered by spontaneous arousals. The rhythmic movements of the body (body rolling) completely resolved following CPAP titration at a setting of 13cm of water along with elimination of breathing events and related arousals. There was one episode of head-rolling during the CPAP titration study. In this section of the study the patient slept for 263min with a sleep efficiency of 98% and a spontaneous arousal index of 15 per hour. The patient felt much better after CPAP titration and the Epworth sleepiness score decreased to 6.

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  • Fig. 1. 

    A 90-s excerpt from a portion of an overnight polysomnography during stage 2 (N2) NREM sleep in a 51-year-old man with upper airway obstructive sleep apnea, restless legs syndrome and rhythmic movement disorder showing two episodes of rhythmic EMG bursts (about 0.75Hz) beginning in the right biceps (Rt Biceps) and right triceps (Rt Triceps) and spreading to other muscles. These bursts were noted following termination of the apneic events and accompanied by arousals and resumption of normal breathing as well as rhythmic body and head-rolling movements (see online video). EEG: top 6 channels (international nomenclature); E1-M1: left electro-oculogram; E2-M1: right electro-oculogram; EKG: electrocardiogram; OralNasal: oronasal thermistor (air flow); PFlow: nasal pressure recording for airflow; Chest: thoracic breathing effort; ABD: abdominal breathing effort; SaO2: oxygen saturation by finger oximetry; Snore: snoring recording; Masseter: right masseter EMG; Chin: submental EMG; RtSterno: right sternocleidomastoideus EMG; LtBiceps: left biceps EMG; LtTriceps: left triceps. EMG; RtBiceps: right biceps EMG; RtTriceps: right triceps EMG; RtQuad: right quadriceps (biceps femoris) EMG; LtQuad: left quadriceps EMG; RtTib: right tibialis EMG; RtGast: right gastrocnemius EMG; LtTib: left tibialis EMG; LtGast: left gastrocnemius EMG.

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  • Fig. 2. 

    A 180-s excerpt from a portion of an overnight PSG during stage 2 (N2) NREM sleep in a 51-year-old man with obstructive sleep apnea, RLS and RMD showing three episodes of rhythmic EMG bursts (about 0.75Hz) and a dystonic EMG burst (last episode towards the end of the tracing) beginning in the right biceps (Rt Biceps) and right triceps (Rt Triceps) and spreading to other muscles. These bursts were noted following termination of the apneic events and accompanied by arousals and resumption of normal breathing as well as rhythmic body and head-rolling movements (see online video). The rest of the figure legend is similar to Fig. 1.

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3. Discussion 

Stereotyped repetitive and rhythmic movements of the body and head occurring predominantly during drowsiness or sleep and accompanied by significant clinical consequences are currently categorized as rhythmic movement disorder [7], [8], [9]. However, videographic recording of these movements during a PSG study has not been adequately documented. The widespread use of video-PSG techniques served to better characterize the known sleep-related motor entities [8], resulting in a variety of previously unrecognized clinical entities (e.g., REM sleep behavior disorder, sleep-related eating disorder, propriospinal myoclonus at the wake-sleep transition). Respiratory-related arousals are associated with increased motor activity, such as respiratory-related PLMS, but the association with other sleep-related motor disorders such as RMD has not been well documented, but briefly alluded to previously (see Figs. 7–2 in Ref. [10]). The pathophysiology of RMD remains speculative. In some patients genetic factors may be involved. Broughton [11] stated that a remarkable feature in a typical rhythmic movement disorder is the absence of any EEG signs of arousal during or immediately after intense rhythmic movements. The recurrence of such rhythmic movements in our patient at the end of the apneic period accompanied by normal breathing and EEG signs of arousal suggest that an arousal may trigger such respiratory-related rhythmic movements in some obstructive sleep apnea patients who are genetically predisposed to an inherent rhythmicity during the sleep-wake transitional stage.

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Appendix A. Supplementary data 

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Supplementary data.

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References 

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PII: S1389-9457(09)00069-0

doi:10.1016/j.sleep.2009.03.003

Sleep Medicine
Volume 10, Issue 4 , Pages 501-503, April 2009

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