Clinical reviewOrthodontics treatments for managing obstructive sleep apnea syndrome in children: A systematic review and meta-analysis
Introduction
Craniofacial growth influenced by genetic inheritance and functional factors can have an impact on general health. Predominant mouth breathing, often caused by increased nasal breathing resistance or adenoid and tonsil hypertrophy, leads to altered muscle recruitment in the nasal and oral cavities, impacting craniofacial growth in a developing child ∗1, ∗2 altering tongue position [3] and oropharyngeal volume, thereby increasing the risk of developing a significant malocclusion. In other words, a small maxilla and/or mandible may predispose children to sleep-disordered breathing (SDB), which is a continuum of severity from snoring to obstructive sleep apnea (OSA).
OSA is a breathing problem occurring during sleep; it is a common chronic disorder in children and adolescents, with a dramatic impact on systemic health [4] and development [5], [6]. Among children and adolescents, the reported prevalence of snoring and OSA is 3–27% and 1–10%, respectively [7], [8], [9], ∗10, [11]. Snoring/OSA is a disorder of upper airway obstruction with multisystem implications and associated complications [11]. Snoring/OSA is often underdiagnosed in children and youth when the primary complaint is a behavioral problem. The American Academy of Sleep Medicine (AASM) states that other problems associated with untreated OSA in children include aggressive behavior [12], attention-deficit/hyperactivity disorder (ADHD) [13] and delays in development [14]. An 11-y longitudinal study on early childhood (4 y old) showed that early sleep problems predicted behavioral and emotional problems in adolescence [15]. If left untreated, OSA can negatively affect a child for the rest of his or her life. There are few proven treatments currently available, and most children are managed with tonsillectomy and adenoidectomy, which have not been demonstrated to fully abolish apnea in all patients, and/or positive airway pressure devices, which have a very poor compliance and are not ideal for all children [16], [17], [18], [19]. Preliminary studies ∗1, ∗2, [3], [15], ∗20, [21], ∗22, ∗23 have suggested that orthodontic treatments, such as maxillary expansion or mandibular advancement with functional appliances, may be effective in handling pediatric snoring and OSA. Accordingly, these preliminary results suggest that the correction of craniofacial structure imbalances during growth may reduce snoring and OSA in children and young adolescents.
This systematic review and meta-analysis focused on two main orthodontic interventions. The first intervention involves an orthopedic mandibular advancement (OMA) that aims to correct dental and skeletal retrognathia by re-directing mandibular growth into a more forward and downward position. This could potentially increase the opening of the oropharyngeal airway during wake and sleep. The second intervention involves rapid maxillary expansion (RME) which is used when the patient is diagnosed with a narrow upper jaw. RME decreases nasal resistance and allows tongue repositioning; as a result, it may reduce the risk of obstruction which contributes to sleep apnea. As a consequence, both interventions hold a probability of becoming valuable alternative treatments for patients who have known craniofacial risk factors, but who are not surgical candidates or are not able to tolerate the standard therapy for OSA or who failed either first-line treatments, i.e. adenotonsillectomy or nocturnal application of positive airway pressure.
Radiological studies indicate that a long and narrow face, a transverse deficiency, and retrognathia are craniofacial morphological factors associated with a narrow upper airway and SDB in children [24], [25], [26], [27]. A recent study found that, compared to obesity, craniofacial morphology was a stronger risk factor for pediatric SDB [28]. Correction of craniofacial risk factors, with orthodontic treatments such as OMA and RME, in optimal conditions afforded by childhood growth may reduce snoring and OSA in children and young adolescents.
In 1860, RME therapy was first published as an orthodontic correction of maxillary constriction [29]. Thus, there is a great body of literature on RME in the fields of orthodontics and dental medicine. However, this therapy was first linked to SDB, when it was shown to decrease nocturnal enuresis in children, a sign and symptom associated to SDB [30], [31], [32]. RME is currently performed most often using a fixed intra-oral orthodontic appliance, which will be adjusted and worn at all times during the treatment. An expansion of 5–8 mm will be obtained over 30 d, with the expansion screw activated daily by parents (active phase). Following this active phase, the expansion screw will be locked into place for a retention phase of 2–6 mo to allow re-calcification of the palatine suture (retention phase).
OMA was first introduced by Dr Kingley with the “bite-jumping” appliance in 1879 [33]. The OMA encourages mandibular growth in a passive or active manner, while being fixed or removable (worn at night, from 21:00 h to 08:00 h). There are many different types of functional appliances, such as monobloc, activator, Frankel, Herbst, bionator and Twin-block [33]. Expected advancement will be of half cusp to full cusp (Class II; 3–6 mm). The mandibular advancement phase of OMA lasts up to 6–9 mo (depending on patient compliance with the removable appliance) followed by approximately 6 mo of retention. Moreover, some of the OMA appliances can be combined with RME appliances.
Following the Pediatric Dental Sleep Apnea strategic planning meeting in 2012 supported by the Canadian Institutes of Health Research, it was recognized that the level of evidence on orthodontic treatments to manage OSA is unknown. More specifically, the number and quality of non-randomized controlled trials (NRCT), and/or controlled before and after studies involving children and adolescents is undefined. There is a need to create a systematic review that includes meta-analysis components to synthesize the data from several studies. This estimation could overcome the barriers faced by clinicians [34] in applying evidence-based medicine and dentistry. To our knowledge, few systematic reviews and/or meta-analyses about craniofacial pediatric OSA have been published [11], [35], [36], [37], [38], [39]. None has yet reviewed and synthesized the available orthodontic treatments for managing OSA in children and young adolescents (i.e. 18 y old or younger). Therefore, there is a need to create a systematic review and meta-analysis of all the available literature regarding orthodontic treatments, such as OMA and RME, for managing OSA in children and young adolescents.
Section snippets
Objectives
Our aims were to investigate the efficacy of the use of OMA (aim 1) and RME (aim 2) in the treatment of OSA in children and young adolescents.
Methods
Methods of analysis and inclusion criteria were specified in advance and documented in a protocol following Cochrane guidelines [40].
Data synthesis
The participants, interventions and outcomes were judged to be sufficiently similar to ensure meaningful findings; therefore, a meta-analysis for each aim was undertaken for the primary outcome. Pooled data for post-treatment results were used for Pirelli et al., 2012 [46], since they presented results separated between responders and non-responders. Qualitative syntheses regarding the secondary outcomes are summarized in the Results section. As the true effect size varied from study to study,
Search and study selection
A flow diagram of the study identification, screening, eligibility and inclusion is shown in Fig. 1 [50]. A total of 58 articles were identified by using the search strategy and sources listed previously. Seven articles [3], [15], [21], [41], [42], [51], [52] were excluded due to identified duplicate data. After the titles and abstracts had been screened, a total of 21 articles [37], [38], [53], [54], [55], [56], [57], [58], [59], [60], [61], [62], [63], [64], [65], [66], [67], [68], [69], [70]
Discussion
OSA is one of the most common sleep disorders to have received attention in the pediatric population within the past decade. The present review and meta-analysis provide updated information on the efficacy of OMA and/or RME treatments for children and young adolescents diagnosed with OSA using AHI as the primary outcome measurement. It is well known that AHI, measured with a sleep study, is an essential criterion for the diagnosis of OSA, confirming signs and symptoms documented by pediatric
Conclusions
The last decade has been marked with pioneer studies resulting in an overall improvement of patient care and underlining the importance of multidisciplinary management of pediatric OSA. Considering the limited number of included studies ∗1, ∗2, [3], ∗10, ∗22, ∗23, [46], ∗49, the presented orthodontic treatments may be effective in managing pediatric snoring and OSA (Fig. 5). Consequently, their respective results suggest that the correction of craniofacial structure imbalances in the optimal
Conflicts of interest
The authors do not have any conflicts of interest to disclose.
Acknowledgments
The Pediatric Dental Sleep Apnea (PDSA) strategic planning meeting (2012) was supported by the Canadian Institutes of Health Research (PLC-122659). Authors are members of this PDSA network. There is no (internal or external) sources support provided for this systematic review and meta-analysis.
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The most important references are denoted by an asterisk.