INTRODUCTION — Sleep-related movement disorders are characterized by relatively simple, usually stereotyped movements that disturb sleep or its onset . By contrast, other sleep-related behaviors, such as sleepwalking and night terrors, are categorized as parasomnias because they involve complex movements and behaviors during sleep. The presence of nocturnal sleep disruption, daytime sleepiness, or other daytime dysfunction is required for a diagnosis of sleep-related movement disorders.
Sleep-related movements of children and adolescents have diverse etiologies, clinical features, management approaches, and outcomes . The movements may occur at the transition from wakefulness to sleep, in sleep, or during both wakefulness and sleep. Some patterns of movement are physiologic and benign (eg, hypnic starts), while others, such as periodic limb movements, when excessive, may be pathologic. The patient may be aware of the disorder in some instances (eg, restless legs syndrome [RLS]) and may be amnestic in another disorder (eg, in some cases of rhythmic movement disorder).
For many of these disorders, a detailed history from the parent or caregiver and the child are sufficient for the diagnosis and may be supplemented by video recordings of the movements obtained in the home environment. In some instances, consultation with a sleep specialist and nocturnal polysomnography are required.
This topic review provides an overview and a clinical approach to sleep-related movement disorders in children. Related topics that address movements and behaviors during sleep include:
CLASSIFICATION — The International Classification of Sleep Disorders, Third Edition, Text Revision (ICSD-3-TR) includes the following categories of sleep-related movement disorders that occur during childhood (table 1) :
●Restless legs syndrome (RLS)
●Periodic limb movement disorder (PLMD)
●Nocturnal muscle cramps
●Sleep-related rhythmic movement disorder
●Benign sleep myoclonus of infancy
●Propriospinal myoclonus at sleep onset
●Isolated symptoms and normal variants
•Sleep starts (hypnic jerks)
•Hypnagogic foot tremor and alternating leg muscle activation
•Excessive fragmentary myoclonus
Disorders from other categories that will be discussed in this topic are:
●Catathrenia (sleep-related groaning) – Classified as a sleep-related breathing disorder in ICSD-3-TR
●Sleep-related epilepsy – Classified as a sleep-related neurologic disorder in ICSD-3-TR
Each of the above will be discussed in this topic review. RLS and PLMD are discussed in more detail in a separate topic review. (See "Restless legs syndrome and periodic limb movement disorder in children".)
Other sleep-related phenomena are categorized as parasomnias because they involve complex movements and behaviors. Common parasomnias in children include disorders of arousal from non-rapid eye movement (NREM) sleep (confusional arousals, sleepwalking, and sleep terrors (table 2)), nightmares, sleep enuresis, sleep paralysis, and rapid eye movement (REM) sleep behavior disorder (which is rare in children). These parasomnias are discussed in a separate topic review. (See "Parasomnias of childhood, including sleepwalking".)
EVALUATION — In many cases, the type of sleep-related movement can be identified based upon a focused history and physical examination (table 1). The general evaluation of a child with sleep-related complaints is discussed in a separate topic review. (See "Assessment of sleep disorders in children".)
History — For a child with complaints of movements or behaviors during sleep, key elements of the evaluation are:
●General questions about the child's sleep:
•Time that the child goes to bed and time it takes to fall asleep (sleep latency)
•Number (and cause) of nocturnal awakenings
•Time that the child wakes up in morning and amount of time it takes to get out of bed
•Does the child feel tired or unrefreshed upon awakening in the morning?
•General daytime functioning, including symptoms of attention deficit hyperactivity disorder (ADHD), irritability, and emotional lability
●Details about the behavior that is causing concern:
•Detailed description of the behavior, including:
-Timing relative to sleep onset.
-Duration of each event and number of events per night.
-Nature of the movements (stereotyped versus random) – Stereotyped movements are characteristic in sleep-related movement disorders or epilepsy, whereas more complex and more variable movements are seen in parasomnias.
-Distribution of the movements (focal or generalized).
-Emesis – Emesis is suggestive of seizures.
-Level of consciousness (confused/responsive) during the episode.
-Large-amplitude body movements, such as rolling over side to side, adjusting bed clothes, or falling out of the bed. These movements may suggest a newly proposed entity, restless sleep disorder. (See 'Restless sleep disorder' below.)
•How often the behavior is occurring (eg, once or twice a month, once or twice a week, or nightly).
•Age of onset of the behavior and any changes in frequency over time, including whether the behavior has been present since infancy (primary) or was initially present, then absent for a period, and then present again at a later age (secondary).
•Whether the child has uncomfortable feelings in the limbs or urge to move the limbs around the time of sleep onset that prevent sleep onset or wake the child. Such symptoms are common descriptors of childhood-onset restless legs syndrome (RLS). It is important to make these inquiries with open-ended questions, using age-appropriate vocabulary, and to have the child describe the symptoms in his or her own words. Young children may describe the symptoms as "owwies," "ouchies," or "feeling like bugs or creepy crawlies are crawling on the legs or arms." (See "Restless legs syndrome and periodic limb movement disorder in children", section on 'History'.)
•Whether there are other sleep-related behaviors, including parasomnias (sleepwalking, confusional arousals, or night terrors).
•Is the caregiver able to provide video documentation of the behavior?
•Does the behavior bother the child or is it bothersome only to the person witnessing the behavior?
●Child's medical history, especially of neurologic disease (eg, epilepsy), delayed developmental milestones, and autism spectrum disorder.
●Child's prescribed medications, over-the-counter medications, and any ingested or topical supplements.
●Family history of epilepsy or sleep-related behaviors. As an example, sleepwalking is often familial.
Home video recording — When possible, we encourage parents and caregivers to provide video recordings of the child's behavior occurring in the home environment. A video may aid in diagnosis, especially when polysomnography in a sleep laboratory is not available or delayed.
Physical examination — Perform a detailed physical examination, including a complete neurologic evaluation, and an assessment for features that are associated with sleep apnea, including craniofacial anomalies like mandibular recession or maxillary and midfacial hypoplasia, tonsillar enlargement, and crowded oral airway. The possibility of obstructive sleep apnea (OSA) is relevant to some movement disorders because OSA can be associated with disorders of arousal from non-rapid eye movement (NREM) sleep, such as sleepwalking and sleep-related bruxism.
Further assessment — The steps above should give a provisional diagnosis. If a specific pathologic disorder is suspected, further evaluation is performed, as needed. As an example, a patient with frequent bruxism should have a dental evaluation.
In some cases, such as an unclear diagnosis or suspected sleep-related epilepsy, consultation with a sleep specialist or neurologist and nocturnal polysomnography, routine electroencephalography (EEG) and/or 24-hour video-EEG telemetry as an inpatient may be required. If nocturnal polysomnography is performed, additional limb leads and expanded EEG montage may be worthwhile for improved characterization of events.
SLEEP-RELATED MOVEMENT DISORDERS — Sleep-related movement disorders are characterized by relatively simple, sometimes stereotyped movements that disturb sleep or its onset . The simple and stereotyped nature of the movements distinguishes them from the complex movements associated with parasomnias (eg, sleepwalking) (table 1). Infrequent manifestations of these movements usually are benign variants, but they are classified as movement disorders when they are frequent or have pathologic consequences.
Restless legs syndrome — Restless legs syndrome (RLS) is a sensorimotor disorder in which patients complain of a sensation of a strong urge to move their limbs, which occurs when at rest and exclusively or predominantly in the evening or at night. Children in comparison with adults may use a wide variety of expressions, terms, or be able to depict their discomfort on drawings to describe this underlying complaint. The diagnostic criteria are shown in the table (table 3). RLS commonly interferes with sleep in both children and adults and is sometimes confused with "growing pains." (See "Growing pains".)
A majority of children with RLS also have increased repetitive limb movements during sleep (known as periodic limb movements of sleep [PLMS], a finding established on nocturnal polysomnography). Periodic limb movement disorder (PLMD) is characterized by more than five limb movements per hour during polysomnography . PLMS tend to occur during non-rapid eye movement (NREM) sleep. Approximately one-third of children with RLS also have associated daytime inattentiveness or hyperactivity .
RLS or PLMD are often associated with iron deficiency and serum ferritin levels below 50 mcg/dL . Iron deficiency can be screened by checking a serum ferritin, while recognizing that ferritin is an acute phase reactant and may be falsely normal or elevated in some cases. RLS/PLMD may also be triggered or exacerbated by insufficient sleep, caffeine, nicotine, and some medications, including sedating antihistamines and serotonergic antidepressants. These disorders are discussed in detail in a separate topic review. (See "Restless legs syndrome and periodic limb movement disorder in children".)
Restless sleep disorder — Restless sleep disorder (RSD) is a proposed diagnostic entity characterized by large muscle group movements of the arms, legs, trunk, or head that are exclusively sleep related, occur most nights, and cause clinically significant impairment (algorithm 1). Video-polysomnography is required for diagnosis .
Although there is restless sleep, the excessive periodic limb movements that characterize RLS are lacking. However, as is seen in RLS and PLMD, patients with RSD may also have concurrent iron deficiency and respond to iron supplementation. (See "Restless sleep disorder in children".)
Nocturnal muscle cramps — Nocturnal muscle cramps are sudden and intense involuntary contractions of muscles (often in the calf or small muscles of the foot) that tend to occur during sleep or while in bed. They may be idiopathic or secondary to other medical conditions. This condition has been best characterized in adults.
In children, they tend to be benign and self-limited. In a retrospective evaluation of 2527 children evaluated in an outpatient clinic, nocturnal leg cramps were diagnosed in 185 children (7.3 percent) . They were only identified in children ≥8 years old, with a peak incidence between 16 and 18 years old. Episodes tended to occur infrequently (one to four times a year) and were most often unilateral, out of sleep, and lasting less than two minutes. (See "Nocturnal leg cramps".)
Sleep-related bruxism — Bruxism is characterized by repetitive clenching or grinding of the teeth and/or bracing or thrusting of the mandible in sleep. It may occur in any of the three stages of NREM sleep (stage N1, N2, or N3). It is most common during childhood, and the prevalence decreases gradually with advancing age. Risk factors include comorbid sleep disorders, especially obstructive sleep apnea (OSA); anxiety and other psychiatric and neurologic disorders; and certain medications and substances, including selective serotonin reuptake inhibitors (SSRIs) and neuroleptic agents (table 4). Bruxism occurs with increased frequency in patients with attention deficit hyperactivity disorder (ADHD) [8,9], and it is unclear whether this is an independent association, or one that is mediated by the medications used to treat the disorder (eg, stimulants). (See "Sleep-related bruxism (tooth grinding)", section on 'Risk factors'.)
Typically, bruxism that occurs infrequently does not need treatment. Bruxism that occurs frequently (eg, nightly) and is severe may warrant treatment with dental devices, such as occlusal splints, that limit wear and tear on the teeth. In particular, children and adolescents with neurodevelopmental disorders or autism spectrum disorder may experience both wakeful and sleep-related bruxism. The severity of bruxism in this population may warrant intervention, although supportive evidence for various therapies is limited. Approaches may include prosthodontics, oral surgery, behavioral modification, music therapy, massage, or even botulinum toxin injections . Additionally, treatment of OSA with adenotonsillectomy may improve the bruxism in patients with or without a neurodevelopmental disorder . The diagnosis and management of sleep-related bruxism is discussed separately. (See "Sleep-related bruxism (tooth grinding)".)
Rhythmic movement disorder — Rhythmic movements of the head, neck, and trunk associated with sleep are common and likely developmental (physiologic) in infants and young children. The term rhythmic movement disorder is used if the movements have or are likely to have significant consequences, such as self-injury or interference with normal sleep . In a study with objective documentation, characteristic rhythmic movements were noted in approximately 1 percent of one-year-old children (maximum prevalence 3 percent) , although much higher rates are reported in studies that rely on parent or caregiver reports. The movements generally resolve spontaneously by five years of age. Rarely, they persist or begin later in childhood or adulthood. Persistence of these behaviors is more common in individuals with neurodevelopmental disorders including ADHD and specific learning disabilities [13-17]. Most studies in children have found no sex difference .
The most common manifestations are body rocking, head rolling (side to side), or head banging (also known as jactatio capitis nocturna), sometimes accompanied by rhythmic humming or inarticulate sounds. Less common are rolling movements of the body or legs or leg banging. In most cases, the child has no recollection of the movements; they are more of a concern to caregivers, who may bring up the behavior with the clinician. Most episodes occur at sleep onset and during NREM sleep (especially stages N1 and N2), but a substantial minority occur during rapid eye movement (REM) sleep . The rhythmic movements most likely represent a release from the descending, suprasegmental inhibition that is normally exercised by the cerebral cortex .
Sleep-related rhythmic movements should be distinguished from the motor stereotypies of children with neurodevelopmental disabilities, such as autism spectrum disorder, which may occur during both wakefulness and sleep. There is no satisfactory treatment, and the movements may gradually resolve spontaneously over time. In cases of forceful head banging, extra padding of the sides of the crib may be required to prevent injury to the child.
Benign sleep myoclonus of infancy — This condition is characterized by repetitive myoclonic jerks of the limbs or the trunk during sleep in infants younger than six months of age . The jerks are typically bilateral, symmetric movements of the arms and/or legs. The condition is uncommon and benign and tends to resolve spontaneously.
The phenomenon is probably due to immaturity of inhibitory motor projections from the cerebral cortex to generators of these movements that are located in the brainstem or spinal cord. Features that help distinguish these myoclonic jerks from epileptic events include the absence of autonomic disturbances, myoclonic jerks occurring only while asleep, and a normal electroencephalography (EEG) and neurologic and developmental examination. (See "Nonepileptic paroxysmal disorders in infancy", section on 'Benign myoclonus of infancy'.)
Infants with neonatal opioid withdrawal syndrome (neonatal abstinence syndrome) often have similar myoclonic jerks, and their clinical course may not be benign. (See "Prenatal substance exposure and neonatal abstinence syndrome (NAS): Management and outcomes".)
Propriospinal myoclonus at sleep onset — Propriospinal myoclonus (PSM) of sleep onset is typically a benign condition characterized by sudden myoclonic jerks that typically occur during transition from wakefulness to sleep . More rarely, the myoclonus occurs during sleep when there are awakenings or with awakening in the morning. PSM has been mainly documented in adults. The jerks are usually flexor movements arising from the trunk, but they can be extensor. Vocalization during events is not common.
Polysomnography with extended EMG recording, including multiple myotomes, may demonstrate that the jerks develop in spinal innervated muscles and then extend caudally and rostrally. Simultaneous EEG recording typically demonstrates a normal alpha rhythm, consistent with relaxed wakefulness, and should not show any epileptiform discharge. MRI of the spine is typically normal; however, pathology is identified in 20 percent of cases. Imaging should be considered when PSM persists during the day. Predominant or exclusive myoclonic jerks occurring with waking in the morning or occurring in clusters should prompt further evaluation for myoclonic seizures. (See "Juvenile myoclonic epilepsy".)
ISOLATED SYMPTOMS AND NORMAL VARIANTS
Sleep starts (hypnic jerks) — These are abrupt, startle-like movements that occur as one is drifting off to sleep, sometimes accompanied by a sensation of falling, a dream-like feeling, or a flashing sensation [1,2]. They are physiologic and are common at all ages. Most of the jerks are isolated, but in children with neurodevelopmental disabilities, they may occur in clusters . Their frequency may be increased by caffeine or other stimulants, prior intense physical exercise, sleep deprivation, or emotional stress.
Hypnic jerks might be mistaken for seizures, but usually can be distinguished clinically. If an electroencephalogram (EEG) is performed, the findings are normal. No treatment is needed for hypnic starts as they are a benign, physiologic entity.
Hypnagogic foot tremor and alternating leg muscle activation — Hypnagogic foot tremor is a rhythmic movement of the feet or toes, occurring around the time of sleep onset with a periodicity of once every one to two seconds . It may involve one or both feet and may last several minutes . The patient may perceive a sensation of movement of the feet. This is a benign entity that is seen in both children and adults. In a study of 375 consecutive patients undergoing polysomnography (adults and children), hypnic foot tremor was documented to occur in 28 subjects (7.5 percent) . The condition may come to attention during the course of investigation for other sleep disorders such as restless legs syndrome (RLS). The simultaneously obtained EEG is normal, thus excluding the possibility of seizures. There are no known adverse consequences, and the disorder likely resolves spontaneously over time.
Alternating leg muscle activation (ALMA) is characterized by an alternating pattern of leg muscle (tibialis anterior) activation on the nocturnal polysomnogram, in which 1 to 2 Hz muscle activations lasting 0.1 to 0.5 seconds are organized into sequences lasting up to 20 seconds. ALMA is likely generated at the level of the spinal cord and results from a loss of descending motor dopaminergic inhibition that is normally exercised on the spinal cord. It may occur with use of selective serotonin reuptake inhibitors (SSRIs) and in conjunction with obstructive sleep apnea (OSA) and periodic limb movements of sleep (PLMS). There may be simultaneous accelerations in heart rate. The cause and management of ALMA remain unknown [22,23].
Excessive fragmentary myoclonus — Excessive fragmentary myoclonus (EFM) is characterized by surface electromyography (EMG) evidence of movement of the fingers, toes, or mouth during sleep, with or without corresponding small visible movements [24,25]. It is a polysomnographic finding that is frequently incidentally identified. EFM is a non-rapid eye movement (NREM) phenomenon that has a clinical appearance that is similar to what is observed with phasic REM twitches; however, EFM is much less common.
EFM is considered a benign, nonprogressive condition. It can occur independently or in association with other sleep problems. It has no known adverse consequences, and whether it contributes to chronic sleep fragmentation remains uncertain.
DISORDERS FROM OTHER CATEGORIES
Catathrenia — Catathrenia (also known as sleep-related groaning) is characterized by a deep inspiration, followed by a prolonged, low-pitched groaning noise [26,27]. There may be several episodes on a given night. Sleep is not disrupted, and the condition is more bothersome to the caregivers or others in the household than to the patient. Because of the prolonged expiration, this is categorized as a sleep-related breathing disorder, but there is no oxygen desaturation or hypoventilation . Video monitoring is able to successfully capture these events. Most episodes occur in rapid eye movement (REM) sleep, but they occasionally occur during non-REM (NREM) sleep. The disorder is benign and there is no satisfactory treatment. (See "Approach to abnormal movements and behaviors during sleep", section on 'Vocalization'.)
Sleep-related epilepsy — There is a close relationship between sleep and epilepsy. Sleep tends to facilitate epileptiform discharges and seizures, perhaps because sleep is associated with synchronized electroencephalography (EEG) patterns. Conversely, epilepsy, antiepileptic drugs, or both may also disrupt sleep quality, such that the prevalence of sleep problems in patients with epilepsy is approximately twice that of the general population (38 versus 18 percent) . Further, patients with epilepsy and their relatives show a higher prevalence of arousal (NREM) parasomnias as compared with controls (odds ratio [OR] 6.3 and 4.7, respectively) .
Sleep-related epilepsy syndromes that occur generally in the absence of daytime seizures include (see "Sleep-related epilepsy syndromes"):
●Self-limited epilepsy with centrotemporal spikes (SeLECTS; also known as [benign] childhood epilepsy with centrotemporal spikes) is the most common of these three entities. The disorder has a peak incidence between seven and nine years of age, and seizures occur only during sleep in approximately 80 percent of patients. The seizures may occur at random times of the night. The child awakens with twitching movements of one side of the face. Consciousness is preserved. Left hemispheric onset of the seizure may be associated with "ictal aphasia." Neurodevelopment is normal, as is the neurologic examination. The disorder tends to resolve spontaneously over time. (See "Self-limited focal epilepsies of childhood", section on 'Self-limited epilepsy with centrotemporal spikes (SeLECTS)'.)
●Sleep-related hypermotor epilepsy and sleep-related temporal lobe epilepsy are uncommon sleep-related epilepsy syndromes that tend to begin during adolescence. They are notable because they are sometimes difficult to distinguish clinically from parasomnias or other sleep-related movement disorders. Features that help to distinguish these disorders from other nocturnal paroxysmal disorders are outlined in the table (table 5).
•In sleep-related hypermotor epilepsy (SHE), previously known as nocturnal frontal lobe epilepsy, associated movements may include paroxysmal arousals, dystonic limb posturing, or hypermotor behavior, which may be in the form of kicking, thrashing, or pedaling movements that may resemble disorders of arousal, nightmares, or panic attacks [30,31]. However, a key distinguishing feature of SHE, as opposed to other diagnostic considerations, is the highly stereotyped nature of the specific movements from episode to episode. The seizures tend to occur randomly through the night and have abrupt onset and offset, with duration generally of only a few seconds. This is in contrast with parasomnias, such as sleepwalking or confusional arousals, which may last two to five minutes and are somewhat more likely to occur in the first third of the night. However, in some patients, SHE events may be longer and include ambulatory behavior that may be difficult to distinguish clinically from sleepwalking. The disorder can be genetic (autosomal dominant) or neurodevelopmental.
•In nocturnal focal epilepsies originating from over the mesial temporal lobe, insula, orbito-frontal lobe, or occipital cortex, there may be automatisms and altered behavior mimicking confusional arousals, ictal vomiting, or nocturnal wandering that resembles sleepwalking. In some cases, there is aura of an unusual epigastric sensation, choking sensation, or retching sensation or a sense of déjà vu. Sleep-related epilepsy with autonomic features is generally idiopathic, in which case, it is sometimes referred to as Panayiotopoulos syndrome . Structural lesions such as malformations of cortical development may, however, underlie the epilepsy in 10 to 20 percent of patients .
Details about sleep-related epilepsy are discussed separately. (See "Nonepileptic paroxysmal disorders in children", section on 'Sleep disorders' and "Sleep-related epilepsy syndromes".)
SOCIETY GUIDELINE LINKS — Links to society and government-sponsored guidelines from selected countries and regions around the world are provided separately. (See "Society guideline links: Parasomnias, hypersomnias, and circadian rhythm disorders" and "Society guideline links: Restless legs syndrome".)
●Definition – Sleep-related movement disorders are characterized by relatively simple, usually stereotyped movements that disturb sleep or its onset (table 1). The simple and stereotyped nature of the movements distinguish them from the complex movements associated with parasomnias (eg, sleepwalking). Infrequent manifestations of these movements usually are benign variants, but they are classified as movement disorders when they are frequent or have pathologic consequences. (See 'Classification' above.)
●Evaluation – Evaluation of a child with concerning or problematic sleep-related movements starts with a careful description of the behavior and a history of sleeping and waking function. In some cases, the diagnosis can be facilitated by video recordings of sleep at home and occasionally requires assessment in a sleep laboratory with polysomnography. (See 'Evaluation' above.)
•Restless legs syndrome (RLS) is a sensorimotor disorder that causes a sensation of a strong urge to move the limbs, mainly when at rest and exclusively or predominantly in the evening or at night (table 3). Children may use a wide variety of expressions, terms, or pictures to describe this sensation. RLS is a common cause of sleep disruption in both children and adults. (See 'Restless legs syndrome' above and "Restless legs syndrome and periodic limb movement disorder in children".)
•Rhythmic movements of the head, neck, and trunk associated with sleep are common and likely developmental (physiologic) in infants and young children. The most common manifestations are body rocking, head rolling (side to side), or head banging, sometimes accompanied by rhythmic humming or inarticulate sounds. The episodes occur in up to 60 percent of nine-month-old infants and generally resolve spontaneously by five years of age. The term rhythmic movement disorder is used if the movements have or are likely to have significant consequences, such as self-injury. (See 'Rhythmic movement disorder' above.)
•Sleep starts (hypnic jerks) are common at all ages and are considered normal. They tend to occur at the time of sleep onset. (See 'Isolated symptoms and normal variants' above.)
●Differential diagnosis – Occasionally, sleep-related epilepsy syndromes can mimic sleep-related movement disorders or parasomnias. In particular, sleep-related hypermotor epilepsy (SHE) may include complex but highly stereotyped movements, such as paroxysmal arousals, dystonic limb posturing, or hypermotor behavior, such as kicking, thrashing, or pedaling movements. (See 'Sleep-related epilepsy' above and "Sleep-related epilepsy syndromes".)
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