Preliminary short-term outcomes of rapid maxillary expansion on periodic limb movements during sleep in pediatric sleep-disordered breathing (2023)


Sleep-related respiratory problems are among the major concerns of parents and caregivers. [1] Up to 50% of children can have a sleep disturbance. Sleep is essential to everyone's routine and dynamic process affecting growth, development, and overall health. [2] Children aged 6–12 years sleep between 9 and 12h per night. Sleep development is related to physical, behavioral, and neurological development, and critical reciprocal relationships exist among these aspects of development. Shortened and poor-quality sleep can lead to high blood pressure, obesity, and depression.

Early identification of sleep problems can prevent negative consequences such as daytime sleepiness, irritability, behavioral and cognitive problems, learning difficulties, and poor academic performance. [3] Fragmented sleep has been associated with neurocognitive and behavioral deficits, including inattention and hyperactivity symptoms. [4,5] In school children aged 6–12 years, the prevalence of sleep problems is about 37%, with 15–25% experiencing bedtime resistance, 10% experiencing delayed sleep onset and anxiety, and 10% experiencing daytime sleepiness. The most common sleep disorders in children are primary snoring, obstructive sleep apnea (OSA), insufficient sleep syndrome, unhealthy sleep habits, sleep enuresis, sleepwalking and night terrors, bruxism, restless legs syndrome (RLS), and periodic limb movement disorder (PLMD). [6]

Periodic limb movements in sleep (PLMS) were first recognized by Symonds in 1953 and were recorded on a polysomnogram by Lugaresi et al., in 1965. [7] PLMS are characterized by repetitive movement of the upper or lower extremities and usually consist of extension of the big toe, partial flexion of the ankle, knee, and hip. Up to 80% of patients with RLS have PLMS. PLMS is considered a diagnostic support criterion for RLS in adults and an essential criterion for children. [8] The polysomnographic documentation of PLMS was a crucial step in supporting the organic nature of RLS. RLS and PLMD are common childhood neurological disorders affecting 2–4%, usually underdiagnosed. [[9], [10], [11], [12]] RLS occurs in 1 in 120 school-aged children. The diagnosis is not even suspected in many children, as these children present with atypical symptoms and associated comorbid conditions. [13] Although about 25% of adults with RLS report the onset of symptoms between 10 and 20 years of age, literature about prevalence in children and adolescents is limited. [14] PLMS is characterized by jerky, repetitive, highly stereotyped limb movements during sleep. These are more commonly seen in the legs, feet, and toes than in the upper extremities.

Some factors can increase the chances of being diagnosed with PLMD and can worsen the PLMD symptoms, including OSA, which causes shallow breathing or pauses during sleep due to an obstruction or block in the throat. The occurrence of one or more obstructive airway events per hour (OAHI) during polysomnography (OAHI ≥1) is diagnostic of OSA. [15] OSA occurs in 1%–5% of children. [16] Snoring can occur without sleep apnea (primary snoring), but a complete evaluation, including polysomnography, should be considered before making this diagnosis. Importantly, there is growing evidence that even primary snoring can have neurodevelopmental consequences. [17]

Adenotonsillectomy (AT) is the primary surgical treatment for OSA in children with adenotonsillar hypertrophy with moderate-to-severe OSA syndrome (apnea-hypopnea index (AHI)≥5) aged >2 years old. [18] However, the surgical approach does not entirely treat pediatric OSA in approximately half of the cases. [19] OSA may recur (or persist) in children with underlying risk factors such as obesity and craniofacial disharmonies.

Rapid maxillary expansion (RME) is an orthopedic procedure that treats OSA-related problems in children with maxillary transverse deficiency (MTD). [20] A transverse deficiency of the maxilla often manifests as a crossbite of the posterior dentition, and can include primary or permanent molars, as well as canines and premolars if severe with a prevalence of 15% in pediatric OSA. [21] Several studies have shown the short-term effectiveness of orthodontic treatment with RME with evidence of significant improvement in OSA. [[22], [23], [24], [25]] RME produces a significant reduction in AHI and a minimal increase in SaO2 immediately after active treatment, at 6–12 months after the beginning of treatment. [26] RME can also improve breathing and decrease snoring rates and the Quality of life (QOL) of children with refractory SDB after AT. [27] Current evidence [28,29] has shown that QOL is a confident prediction of behavioral outcomes.

Sleep-related movement disorders are an important group of sleep diseases encountered commonly in pediatric clinical practice. Few studies have assessed PLMS treatment outcomes in children with sleep-disordered breathing (SDB). Knowing that some children needed another treatment modality according to their diagnosis, this study aimed to evaluate the outcome of RME on PLMD in children with maxillary constriction and residual snoring post-AT.

Section snippets

Study design, recruitment, and data collection

Patients with residual snoring and MTD who had undergone adenotonsillectomy for at least two years were selected for this prospective clinical trial. These patients were supervised and treated at the Pediatric Otorhinolaryngology Outpatient Clinic of the Escola Paulista de Medicina of the Universidade Federal de São Paulo (UNIFESP). This orthodontic intervention study was registered in the Brazilian Registry of Clinical Trials (ReBEC): RBR-463 by.

For this study we initially contacted by


The sample comprised 24 white Brazilian patients of mixed ethnic ancestry, aged between 6.1 and 12.7 years before RME. The mean age was 10.0 years, and the standard deviation was 1.8 years. On average, they weighed 36.0kg and were 1.36m high. As expected, age, weight, and height significantly increased between the Pre-RME and Post-RME periods (p<0.001) (Table 1). As for age-adjusted BMI (Z-score for age), the mean decreased from 0.87 (SD=2.16) before RME to 0.37 (SD=1.76) after RME,

(Video) Early Identification and Treatment for Pediatric Sleep Disordered Breathing with Stacy Ochoa, DDS


Childhood sleep disorders are common and associated with significant impairment in QOL. [38] The most crucial finding in this study was the association between the improvement of QOL of these children with sleep disorders and the PLMS. Problems with sleep are relatively common but can often be challenging to recognize. When sleep-deprived, children and adolescents are often openly moody. [[39], [40], [41]]

To the best of our knowledge, this is the first study evaluating the effects of RME on


Our study showed a significant improvement in the PLMS presented in the polysomnography after RME treatment. The improvement in PLMS in the OSA group suggests a positive neurological impact of the treatment. RME may be an alternative treatment to improve the QOL in children with SDB and MTD. We suggest a multi-professional approach to the treatment of sleep disorders in children.


CAPES: Coordenação de Aperfeiçoamento de Pessoal de Nível Superior.

AFIP: Associação Fundo De Incentivo a Pesquisa.

Declaration of competing interest

None declared.


We would like to thank Thais Moura Guimarães for her advice and help in collecting research data.

Thanks to Associação Fundo de Incentivo à Pesquisa – AFIP.

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    The present prospective clinical study aimed to investigate the effects of rapid maxillary expansion on the airway, correlating airway volumes obtained on multi-slice computed tomography and polysomnography assessment of oxygen saturation and apnea/hypopnea index.

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What are periodic limb movements in sleep PLMS? ›

Periodic limb movement disorder or PLM is also referred to as sleep-related myoclonus syndrome or nocturnal myoclonus syndrome. These are movements of the legs or upper limbs during sleep, that are periodic, and have stereotypic behavior.

What are the symptoms of periodic limb movement disorder? ›

What Are the Symptoms of PLMD? The main symptoms of periodic limb movement disorder include poor sleep, daytime sleepiness, frequent awakenings, and rhythmic movements involving one or both legs during sleep.

What stage of sleep does periodic limb movement occur? ›

PLMS are most frequent during non-rapid eye movement (NREM) sleep stages 1 and 2. The movements become less frequent during stage 3 of NREM sleep and during REM sleep.

What causes sudden leg movement while sleeping? ›

Restless legs syndrome (RLS) is a condition that causes an uncontrollable urge to move the legs, usually because of an uncomfortable sensation. It typically happens in the evening or nighttime hours when you're sitting or lying down. Moving eases the unpleasant feeling temporarily.

Is PLMD a neurological disorder? ›

Periodic limb movement disorder (PLMD) is one of the commonest neurological disorders and causes significant disability, if left untreated. However, it is rarely diagnosed in clinical practice, probably due to lack of awareness and/or lack of necessary diagnostic facilities.

How do you treat PLMD in children? ›

How are RLS and PLMD treated? Patients with RLS or PLMD are treated with a combination of learning new behavioral skills, vitamin supplements and other medication. Good sleep habits: It is important for children and adolescents to get enough sleep and have a regular sleep schedule.

Is periodic limb movement disorder a VA disability? ›

Since the award of service connection, an initial rating of 10 percent for periodic limb movement disorder, but no more, is granted, subject to the law and regulations governing the payment of monetary benefits.

What is the best treatment for periodic limb movement disorder? ›

Clonazepam (Klonopin), in particular, has been shown to reduce the total number of periodic limb movements per hour. It is probably the most widely used drug to treat PLMD.

Is periodic limb movement disorder the same as restless legs syndrome? ›

Restless leg syndrome and periodic limb movement disorder have similar symptoms and often occur together, but they are distinct conditions that require different tests and sometimes different treatments.

Is PLMD related to Parkinson's? ›

PLMS are very common in patients with narcolepsy, REM behavior disorder, and Parkinson's disease. True PLMD – the diagnosis of which requires periodic limb movements in sleep that disrupt sleep and are not accounted for by another primary sleep disorder including RLS – is uncommon.

How is periodic limb movement disorder diagnosed? ›

How do I know if I have periodic limb movement disorder (PLMD)? The diagnosis is based on the clinical history as well as an overnight polysomnogram (PSG). This is a test that records sleep and the bioelectrical signals coming from the body during sleep. A thorough neurological examination should be performed.

Does melatonin help PLMD? ›

Medications that have been shown to help in small studies of people with PLMD include: Melatonin. Clonazepam (Klonopin) Valproate.

Is periodic limb movement disorder serious? ›

Primary PLMD is not considered medically serious, although complications arising from the condition may cause issues. According to the National Sleep Foundation, primary PLMD is uncommon.

What drugs cause REM sleep behavior disorder? ›

RBD has been associated with antidepressant medications such as tricyclic antidepressants, fluoxetine, venlafaxine, and MAO inhibitors. Although REM behavior disorder has been associated with the use of serotonergic reuptake inhibitors, there are actually very few documented cases in the literature.

What is periodic limb movement disorder neurology? ›

Periodic limb movement disorder (PLMD), also known as nocturnal myoclonus, is a condition where leg muscles contract and jerk every 20 to 40 seconds during sleep. The movement in the leg is the extension of the big toe, while at the same time the ankle, knee and sometimes the hip are partly flexed.

What is the most common neurological movement disorder? ›

The most common movement disorders are essential tremor, restless legs syndrome, and Parkinson's disease. Movement disorders range from mild to severely debilitating, and many have very similar symptoms. It is vitally important to get an accurate diagnosis.

What disease is associated with movement disorder? ›

Parkinson's disease and certain dopamine blocking medications are the most common causes. Other causes include degenerative disorders such as multiple system atrophy and progressive supranuclear palsy.

What is the first line treatment for periodic limb movement disorder? ›

Gabapentinoids — Pregabalin, gabapentin, and gabapentin enacarbil are all appropriate first-line therapies for chronic persistent RLS in many patients [7,49,50].

Can PLMD be caused by anxiety? ›

PLMD is also known to be associated with depression and anxiety disorder [10].

What is severe periodic limb movement disorder? ›

Periodic limb movements are when you have episodes of simple, repetitive muscle movements. You are unable to control them. They usually do not keep you from falling asleep. Instead, they can disrupt your or your bed partner's sleep during the night. This can cause you both to be very tired during the day.

How much VA disability can you get for MST? ›

A temporary disability rating of 100% is available if a veteran must be hospitalized for 21 days or more due to their mental health condition. Treatment must be received at a VA medical center or a VA-approved facility to qualify for the temporary total disability rating.

What is the average VA disability rating for MST? ›

The most common MST-related disability is PTSD which is rated at 10%, 30%, 50%, 70% or 100% based upon a specific listing of symptoms and impact on the veteran's day-to-day life. That rating, then, is matched up to a set of marital/family statuses.

Can magnesium help PLMD? ›

Magnesium supplementation is often suggested for restless legs syndrome (RLS) or period limb movement disorder (PLMD) based on anecdotal evidence that it relieves symptoms and because it is also commonly recommended for leg cramps.

What muscle relaxers are used for PLMD? ›

Gabapentin (also known by its brand name of Neurontin) is the most commonly prescribed anticonvulsant for PLMD. Muscle relaxants: Medications such as baclofen (brand name Lioresal) that inhibit the brain chemicals that stimulate muscle contractions have been shown to lessen the effects of PLMD.

What neurological disorder causes restless leg syndrome? ›

Restless legs syndrome (RLS)—also known as Willis-Ekbom Disease, primary RLS, and idiopathic RLS—is a neurological disorder that causes unpleasant or uncomfortable sensations in your legs and an irresistible urge to move them.

Can gabapentin help restless legs syndrome? ›

Gabapentin enacarbil is used to treat moderate-to-severe primary Restless Legs Syndrome (RLS). RLS is a neurologic disorder that makes the legs feel uncomfortable. This results in an irresistible feeling of wanting to move your legs to make them comfortable.

What are the four cardinal signs of Parkinson's disease? ›

One of the most prevalent neurological disorders is Parkinson's disease (PD), characterized by four cardinal signs: tremor, bradykinesia, rigor and postural instability.

What syndrome mimics Parkinson's? ›

Progressive supranuclear palsy (PSP).

This condition mimics Parkinson's disease most closely early in its progression. Later, unique symptoms develop, including eye movement limitations and trouble swallowing, speaking, thinking, and sleeping.

Is PLMD related to sleep apnea? ›

Periodic leg movements in sleep (PLMS) are found in 24-48% of patients with obstructive sleep apnea (OSA).

Does CPAP help PLMS? ›

Background: Both obstructive sleep apnea (OSA) and periodic limb movements of sleep (PLMS) are common in the sleep laboratory. The severity of OSA can be improved by using continuous positive airway pressure (CPAP).

How many PLMS are normal? ›

There should be at least four leg movements in a 90-s period. Contractions should be more than 0.5-s and less than 5-s.

How serious is PLMD? ›

Primary PLMD is not considered medically serious, although complications arising from the condition may cause issues. According to the National Sleep Foundation, primary PLMD is uncommon.

Is PLMD related to MS? ›

Sleep related disorders most commonly reported in MS are as follows: insomnia, sleep-related breathing disorders (SRBD), restless legs syndrome (RLS) and periodic limb movement disorders (PLMD).

What are the long term effects of PLMD? ›

RLS and PLMD can lead to several long-term consequences. RLS and PLMD can lead to daytime tiredness, sleepiness or hyperactivity. Children may have attention problems and may not be able to do well at school. Some children may be mistakenly diagnosed with ADHD.

What is the difference between PLMD and PLMS? ›

Periodic Limb Movements in Sleep (PLMS)

PLMS is often mistakenly called periodic limb movement disorder (PLMD), which is a disorder when patients with PLMS complain of insomnia or hypersomnia that cannot be explained by other secondary causes. Up to 80% of patients with restless legs syndrome (RLS) have PLMS.

Which is worse apnea or hypopnea? ›

While apneas are generally considered more severe than hypopneas, there is evidence that both carry similar risk factors. View Source for cardiovascular disease and other illnesses.

What is the difference between RLS and PLMD? ›

Periodic limb movement disorder involves repetitive movements of the arms, legs, or both during sleep. Restless legs syndrome involves an irresistible urge to move and usually abnormal sensations in the legs, arms, or both when people sit still or lie down.


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