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Criteria Recommendation
by the quantity and severity of desaturati ons observed in an overnight pulse oximetry
recording (Table 17.2). A score higher than one was proposed as an indication of
obstructive sleep apnea (OSA). They suggested that prioritizing operative interven-
tion based on oximetry score can shorten the diagnostic and treatment process for
those with more severe diseases. However, oximetry is inconclusive in many
children with suspected OSA and cannot repla ce PSG for the definitive diagno sis
of OSA (Nixon et al.
2004)
.
294 C. D’Elia et al.
Table 17.2 The McGill Oximetry Scoring System (MOS) (Nixon et al. 2004)
Oximetry
score
No. of
drops
in
SaO
2
<90%
No. of
drops
in
SaO
2
<85%
No. of
drops
in
SaO
2
<80%
1 Normal
study/incon-
clusive for
OSA
<3 0 0 Baseline: stable
(<3 clusters of
desaturation)
and >95%
Additional evalua-
tion of breathing
during sleep
required to rule out
OSA
2 OSA, mild >3 3 0 Three or more
clusters of
desaturation
events
Recommend T&A
on the waiting list
3 OSA,
moderate
>3 >3 3 Three or more
clusters of
desaturation
events
Recommend surgery
within 2 week
4 OSA, severe >3 >3 >3 Three or more
clusters of
desaturation
events
Recommend urgent
surgery (within
days)
In agreement with other studies (Chan et al. 2019; Chuanprasitkul et al. 2021;
Jonas et al. 2020), it was shown that nocturnal oximetry may not be used for a
definite diagnosis of OSA in obese children (Van Eyck e
t al.
2015). Although it has
good specificity, with a positive result most likely indicating a positive PSG result,
an absence of abnormal oximetry results does not definitively exclude a diagnosis of
OSA, as the sensitivity of this test is only moderate (Van Eyck et al. 2015). In the
context of a child suspected of having OSA, Brouillette et al. reported that positive
nocturnal oximetry results have a minimum positive predictive value of 97% for
OSA, whereas negative oximetry findings cannot completely rule out the possibility
of OSA (Brouillette et al. 2000).
OSA by full night polysomnography was demonstrated in 42% of children who
had a normal or inconclusive McGill oximetry score (MOS 1) (Horwood et al.
2014). Despite the low negative predictive value, the utility of nocturnal oximetry
as a particularly effective tool for identifying cases of severe OSA was verified given
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that the clinical values for this group, such as the AHI, desaturation index, and
oxygen saturation nadir, designate these children as having OSA in the mild to
moderate range and without clinically significant desaturations (Horwood et al.
2014). Additionally, in another study, the specificity of MOS 3 and 4 in diagnosing
moderate/severe OSA on PSG was 100% (Jonas et al. 2020).
17 Updates in Pediatric Sleep Apnea 295
MOS scores over different nights may h ave
only been fair to a moderate agree-
ment. In a recent study of children, 38% screened positive for OSA on the first night.
When three nights of oximetry were done 59% screened positive on at least one of
the nights suggesting that one night of oximetry screening may not be sufficient to
screen for OSA (Galway et al.
2021).
Efforts to develop novel strategies for the analysis of oximetry data have been
heavily concentrated on the development of tools for the automated (or semi-
automated) diagnosis of OSA (Terrill 2020).
17.14 Pediatric OSA Treatment
Due to its complexity, OSAS management requires a multidisciplinary approach by
different specialist teams to make an early diagnosis and a correct treatment plan.
Several specialists are engaged in the treatment of pediatric OSA such as pediatri-
cians, otorhinolaryngologists, pediatric dentists, orthodontists, neurologists, nutri-
tionists, physiotherapists, and speech therapists (Giuca et al.
2021).
Although AH is the most common risk factor for Pediatric OSA, obesity has
become an increasingly important risk factor. Prevalent, especially in early child-
hood (6–9 years) and adolescence (Bue et al.
2020). Therefore, nutritional education
and physical exercise/training may be a part of the OSA therapeutic plan (Giuca et al.
2021).
The size and location of tonsils and a denoids are influenced by genetics, infec-
tion, and inflammation. Although tonsils that look big may contribute to a reduction
in the diameter of the pathways, there is no clear linear correlation between enlarged
tonsils and adenoids with the severity of OSA. Therefore, even the “small” tonsils
(e.g., within the tonsillar pillars) may be clinically significant and cause airway
obstruction during sleep. Adenoids also contribute to the reduction in airway size
and are better visualized by nasal endoscopy or magnetic resonance image
processing and anatomic measurements (Arens and Marcu s 2004; Arens et al. 2001).
Children with OSA experience a combination of oxidative stress, inflammation,
autonomic activation, endothelial dysfunction, increased aggregation platelet count,
metabolic dysregulation, and interruption of sleep homeostasis (O’Driscoll et al.
2009). OSA is a common disorder in children and those at risk must be identified,
studied, and treated promptly because untreated OSA can be responsible for cardio-
vascular, metabolic, and neurocognitive morbidities, with the potential to induce
deficits, sometimes nonreversible, given its persistence in a critical period of phys-
ical and neuropsychic development (Bue et al.
2020). Treatment of OSA should be
viewed as rather urgent in children, particularly when daytime symptoms such as
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hyperactivity, inattention, somnolence, or poor school performance are detected by
either parents or school teachers (Gozal et al. 2021).
296 C. D’Elia et al.
Treatment improves behavior, attention, quality of life,
neurocognitive function-
ing, enuresis, parasomnias, and restless sleep and reversing of associated cardiovas-
cular sequelae (Chinnadurai et al.
2017).
In addition to adenotonsillectomy, the treatment of OSA in children may include
the use of continuous positive airway pressure (CPAP), weight loss, oral devices,
and pharmacological treatment.
The decision to initiate treatment and choice of treatment depend upon the child’s
age, clinical symptoms (e.g., nighttime sleep problems or daytime dysfunction),
presence of comorbidities (especially underlying genetic, craniofacial, neuromuscu-
lar disorders), risk factors (e.g., obesity, crowded oropharynx), and results of PSG, if
performed.
17.15 Medical Therapy
Inflammation is consistently present in adenotonsillar tissues and upper airway
tissues in children with OSA and treatment with corticosteroids and leukotriene
receptor antagonists may reverse the underlying processes and resolve or mitigate
OSA (Gozal et al.
2021; Goldbart and Tal 2008).
Airway inflammation is thought to be caused by the local, repeated, mechanical
trauma related to the vibratory intermittent airway occlusion typical of OSA.
Another potential mechanism mediating local and systemic inflammation involves
the intermittent nocturnal hypoxemia that may, through the phenomenon of the
ischemia-reperfusion injury, produce oxygen-free radicals (Goldbart and Tal
2008).
Corticosteroids are particularly effective in alleviating the magnitude of the
inflammatory processes in the airway via their binding to corticosteroid receptors
and their translocation to the nucleus where they operate via transcriptional, epige-
netic, and post-translational regulatory processes (Gozal et al. 2021). The expression
of cysteinyl-leukotriene receptors is markedly enhanced in the tonsillar and adenoid
tissues of children with OSA and are predominantly located in T cell lymphocytes
(both CD4+ helper T cells and even more so in CD8+ cytotoxic T cell lymphocytes).
In vitro studies using mixed cell cultures of either adenoids or tonsils showed that the
addition of leukotriene D4 to the media resulted in increased cellular p roliferation
and release of pro-inflammatory cytokines. When such cultures were treated with
leukotriene receptor antagonists or with corticosteroids, dose-dependent reductions
in proliferation and cytokine release occurred (Gozal et al.
2021).
Anti-inflammatory agents, especially topical nasal spray corticosteroids, are
suggested to have a potential role in reversing adenotonsillar enlargement (Chan
et al. 2015).
The initial randomized controlled trial (RCT) examining the effectiveness of
nasal fluticasone for a duration of 6 weeks in children diagnosed with OSA who
were waiting for AT was conducted in 2001, with a substantial decrease of the AHI
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in the fluticasone group (Brouillette et al. 2001). In a larger RCT, intranasal
budesonide for 6 weeks in PSG-diagnosed children with mild OSA resulted in
significant improvements in both polysomnographic measures (sleep latency,
slow-wave sleep, and rapid eye movement sleep) and in the magnitude of respiratory
disturbance (apnea/hypopnea index, nadir pulse oxygen saturation). Lateral X-rays
of the upper airway showed that the size of the adenoids had also declined
(Kheirandish-Gozal and Gozal
2008)
.
I
n a more recent RCT of intranasal
mometasone furoate (MF) for 4 months versus placebo in children aged
6–18 years with mild OSA, AHI, and ODI improved significantly in the MF group
only (Chan et al. 2015).
17 Updates in Pediatric Sleep Apnea 297
However, the available evidence regard
ing the effectiveness of intranasal corti-
costeroids in treating OSA in children is insufficient. The level of certainty regarding
the benefits on the primary outcome, including the apnea–hypopnea index (AHI) and
respiratory arousa l index, is considered low due to imprecise estimates and varia-
tions among studies (Kuhle et al.
2020). A large number of studies have been
published on intranasal corticosteroids, but the majority did not assess the children
using PSG (Gozal et al.
2021).
After the initial study in 2005, which showed improvements in AHI in children
with mild OSA in an open-label trial (Goldbart et al. 2005), two double-blind, RCTs
have been conducted and revealed that oral montelukast therapy for 12–16 weeks
improves nocturnal symptoms, reduces the size of adenotonsillar tissues, and sig-
nificantly reduces PSG-derived measures in the context of mild OSA (Goldbart et al.
2012; Kheirandish-Gozal et al. 2016 ).
In a review conducted in 2016, examining the VigiBase databa se analysis of
psychiatric disorders in children linked to the use of Montelukast database,
age-related neuropsychiatric adverse drug events were observed. Infants and chil-
dren were found to develop sleep disturbances, while adolescents were more prone
to experiencing depression/anxiety and psychotic reactions (Aldea Perona et al.
2016). The emergence of side effects related to montelukast raises a red flag that
should prompt caution when deciding to use this compound (Gozal et al.
2021).
Studies comparing head-to-head intranasal corticosteroids and oral montelukast
revealed rather similar improvements in children with OSA. Based on these results,
both therapies seem to be of comparable efficacy (Gozal et al. 2021).
Overall, beneficial effects occurred in >80% of 752 children with combination
therapy with intranasal corticosteroids and oral montelukast. In a subgroup of
445 patients who underwent follow-up polysomnography, 62% demonstrated
improvement and normalization of sleep findings, while 17.1% experienced either
no improvement or a worsening of their OSA. Among those who did not respond
positively, older children (aged >7 years) and obese children (with a BMI z-
score > 1.65) were notably more likely to be nonresponders (Kheirandish-Gozal
et al. 2014).
Bacterial involvement in AH was first suggested based on the increase in lym-
phocytes in tonsils and adenoids and later based on a greater number of bacterial
pathogens such as Haemophilus influenzae and other β-lactamase-producing bacte-
ria in enlarged tonsils and adenoi ds (Don et al.
2005). Some studies have indicated
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the efficacy of broad-spectrum antibiotics to improve the symptoms and complica-
tions of AH and OSA. However, the results of a meta-analysis, which analyzed
various forms of treatment for OSA, found statistical insignificance for all compar-
isons related to antimicrobial therapy (Lin et al.
2020). Evidence of the usefulness of
using broad-spectrum antibiotics to treat pediatric OSA is limited.
298 C. D’Elia et al.
Children with mild or moder
ate OSA and nasal obstruction due to adenoid
hypertrophy, especially those with seasonal allergies, may be candidates for treat-
ment with intranasal corticosteroids or leukotriene antagonist therapy. These anti-
inflammatories are typically considered as an alternative to adenotonsillectomy, or as
a therapy during the watchful waiting period before other interventions. If this
therapeutic option is selected, an administration of at least 4 weeks of the drug
must be performed to decide whether it will be useful to maintain the therapy for a
longer period.
17.16 Adenotonsillectomy (AT)
If a child meets diagnostic criteria for OSA, clinical examination consistent with AH,
and no contraindications to surgery, adenotonsillectomy should be recommended as
the first line of treatment (Marcus et al. 2012). Certain children may present with
multiple degrees of airway obstruction and might necessitate further assessment and
treatment (Bitners and Arens
2020).
A variety of surgical techniques and instruments such as “cold dissection, electric
monopolar cautery, bipolar cautery, harmonic scalpel, bipolar radiofrequency
(coblation), and PEAK PlasmaBlade (Medtronic)” has been used for AH surgery.
Various methods and devices have been employed to decrease post-operative pain
and intra-operative bleeding, increase surgical speed, and decrease the risk of post-
surgical hemorrhage (Sakarya et al.
2017). Adenoidectomy can be performed using
suction diathermy ablation or curettage adenoidectomy (Sakarya et al.
2017).
A multicenter randomized controlled trial (Childhood Adenotonsillectomy
Trial—CHAT) examined the outcome of early adenotonsillectomy compared with
a conservative approach of watchful waiting without surgical intervention
concerning neurobehavioral symptoms, quality of life, and changes in
polysomnography (Marcus et al.
2013
). The study population included
464 school-aged children (5–9 years old) with mild sleep apnea, moderate OSA,
or severe OSA determined by polysomnography. More than 50% of subjects were
African American and nearly 50% were overweight or obese. The primary outcome,
attention, and executive function on the Developmental Neuropsychological Assess-
ment did not show any significant difference in the early tonsillectomy group
compared with the watchful waiting group. However, compared with the watchful
waiting strategy, there was a significant improvement in behavior, reduction of OSA
symptoms, normalization of polysomnography findings, and quality of life (evalu-
ated by the pediatric quality of life inventory) in children undergoing early
adenotonsillectomy. Although the percentage of subjects experiencing
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normalization of polysomnography after early adenotonsillectomy was higher than
that in the watchful waiting group (79.5% vs. 46.5%), it should be noted that almost
half of the subjects in the non-surgical intervention group demonstrated normaliza-
tion of polysomnography at the end of the 7-month follow-up period. This improve-
ment in the non-intervention group was attributed to the growth of the developing
airway leading to a proportional reduction in airway obs
truction because of AH
(Marcus et al.
2013)
.
17 Updates in Pediatric Sleep Apnea 299
A recent systematic review
suggests that AT treatment of OSA in children can
improve behavioral symptoms, cognitive outcomes, and sleep-related quality of life.
The clear improvement of patients after AT provides evidence for a cause-and-effect
relationship between sleep breathing disorders and various negative results on
behavioral, cognitive, and mental health (Di Mauro et al.
2021).
Using a multicenter study design consisting of eight pediatric sleep centers across
the USA and Europe that routinely conduct preoperative and post-operative noctur-
nal polysomnography, the outcomes of adenotonsillectomy for OSA in a large
cohort of children were examined and revealed a high frequency of residual OSA.
Approximately 50% of included children were obese and only 27.2% had complete
resolution of OSA (Bhattacharjee et al.
2010). There is compelling evidence that AT
is not uniformly effective in curing OSA in children and that in the context of
increasing obesity rates in children, the ability of AT to eradicate OSA needs to be
viewed with great skepticism (Bhattacharjee et al.
2010).
The impact of adenotonsillectomy in children with severe OSA without
comorbidities was reviewed (Galluzzi and Garavello 2021). AT significantly reduces
apneas in otherwise healthy children with severe OSA. All the studies included
documented a significant reduction of AHI or RDI values after adenotonsillectomy,
the improving varied from 57.7 to 93.3% (Galluzzi and Garavello
2021). Neverthe-
less, the number of children with severe OSA having persistent apnea after surgery is
high. Considering AHI ≥5, 30 to 55% of children still have apneas and with AHI ≥1
the percentage increases from 60 to 90.6% (Galluzzi and Garavello 2021).
Various risk factors for persistent OSA in otherwise healthy children with severe
OSA have been investigated with contradictory data (Galluzzi and Garavello 2021).
Such factors include small adenoids, abnormal facial morphology, narrower
epipharyngeal airspace, more poorly develo ped mandibular protrusions, OSA sever-
ity, age, obesity, male sex, allergy, and asthma. Overall, to date, the demographic
characteristics and the polysomnographic criteria that will predict residual OSA in
otherwise healthy children after AT have not been completely established. This may
be reflected in the unknown etiology of OSA which is probably due to multifactorial
causes (Galluzzi and Garavello 2021).
Tonsillotomies instead of tonsillectomy are preferred by many surgeons since it
has lower post-operative complication rates such as hemorrhage, less post-surgical
pain, and shorter recovery times (Tan et al.
2016). there are no significant differences
in the resolution of OSA symptoms, quality of life, and post-operative immune
function between the two surgical procedures. However, the risk ratio of OSA
recurrence was 3.33 in favor of tonsillectomy (Wang et al.
2015
). Current data are
insufficient to recom mend one surgical technique over the other. Children
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undergoing tonsillotomy should be monitored carefully long-term to ensure that
OSA symptoms do not recur (Tan et al. 2016).
300 C. D’Elia et al.
Following AT, it is important to moni
tor all children for any lingering symptoms,
and in cases of persistent disease, repeat polysomnogr aphy may be conducted to
identify the need for ongoing management, including positive airway pressure
ventilation and potentially additional surgical interventions if necessary (Bitners
and Arens 2020).
17.17 Continuous Positive Airway Pressure (CPAP)
Treatment with CPAP is provided through an electronic device that maintains
constant positive air pressure through a nasal or face mask, with effects of the
mechanical endoprosthesis of the airways and improvement of functional residual
capacity. The goals of CPAP therapy are to ensure unobstructed breathing, good
sleep quality, improved daytime funct ion, and in some cases, rest from the work of
breathing by overcoming the critical closing pressure of the pharynx. It has no
restrictions or recommendations regarding the age of application.
For patients with specific surgical contraindications, craniofacial abnormalities,
minimal adenotonsillar tissue, persistent OSA after adenotonsillectomy, or for those
who prefer non-surgical alternatives, CPAP therapy is an option (Gao et al.
2021). It
may also be indicated as a bridge to surgery or orthodontic procedures (craniofacial
alterations, severe adenoid, and tonsil hypertrophy with repercussions on growth
and/or cardiac function). However, unlike adenotonsillectomy, which is a single
procedure that, in most cases, results in significant resolution or improvement,
CPAP, in many cases, will need to be used indefinitely (Section on Pediatric
Pulmonology et al.
2002).
CPAP has been increasingly used in the treatment of pediatric OSA unresolved
by surgical airway intervention. It is not clear what proportion of children can be
successfully withdrawn from CPAP and the factors that affect this discontinuation.
In an Australian cohort , reasons for discontinuing CPAP included post-surge ry
improvement in the airway, weight loss, and improvement in symptoms and/or PSG
parameters over time. It was concluded that the natural course of pediatric OSA is
not clear, especially in severe and treatment-resistant cases (King et al. 2019).
Children on CPAP should be monitored regularly for maintenance of symptoms
and the need for positive pressure support, as a small proportion can be successfully
removed from CPAP over time.
Initial CPAP prescriptions are made by specialists in sleep medicine reference
centers, ideally integrated into multidisciplinary consultations that ensure the neces-
sary diagnostic workup for therapeutic decisions.
The pressure levels needed vary between individuals; therefore, CPA P should be
titrated in the sleep laboratory prior to devise prescription and periodically readjusted
thereafter. The auto-CPAP machines are programmed to adjust CPAP pressure
based on the detection of apnea, hypopnea, flow limitation, snoring, or free-flowing
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air. When successful, this precludes the need for CPAP titration in the laboratory.
The pressure applied is set within a range determined by the prescriber The long-
term use of CPAP may cause craniofacial abnormalities; thus, regular evaluation is
recommended (Gao et al.
2021).
17 Updates in Pediatric Sleep Apnea 301
Objective monitoring of adherence, by
means of software data analysis, is
essential. If adherence is less than ideal, measures to improve, such as the behavioral
modification or treatment of side effects (nasal symptoms, optic irritation, and skin
damage) should be instituted. If these measures are ineffective, the instit ution of
alternative treatments should be considered (Marcus et al.
2012).
CPAP should be used only in subjects with obstructive sleep apnea in the absence
of hypoventilation. Bilevel-positive airway pressure (BiPaP) may be used instead of
CPAP for patients with significant hypoventilation (elevated PaCO
2
) or clinically
significant central apneas (Arens and Muzumdar 2010).
CPAP is generally better tolerated in older children. Behavioral or desensitization
techniques may be necessary to help young children or older children with learning
or behavioral difficulties become accustomed to using the device.
17.18 Odontoesthomatological Management
Odontostomatological care providers are likely to be an important part of the sleep
medicine team either in adults or pediatrics (Meira et al. 2020). Although from a
traditional perspective, oral health professional does not make the initial diagnostic
judgment, they can contribute to identifying pediatric OSA as they examine children
and adolescents more frequently than pediatricians or family doctors. Furthermore,
orofacial and craniomandibular-specific features may be strongly associated with
some particular subsets of OSA in these age groups (Stauffer et al.
2018). Still,
associations between OSA and orofacial characteristics are bidirectional, with mor-
phometric changes predisposing to an impaired respi ratory function during sleep
and, breathing difficulties or changes in normal respiratory pattern, determining
skeletal and soft tissue developmental challenges which perpetuate in a vicious
cycle of unless there is an interventive measure to stop this negative feedback
loop. Even though as children get older, other features prevail as etiological deter-
minants for the development and worsening of OSA, this relationship remains of
high relevance in a wide range of pediatric ages.
It became clear from several epidemiological and clinical studies, that phenotypic
rather than anatomic markers regarding craniofaci al features are linked to pediatric
OSA. These markers are for instance long narrow fascia, convex profile, some
degree of constriction within the palate, and anterior open bite. However, they
seem to play only a minor role in other subsets of patients.
Therefore, as the positive predictive value of those markers is high, it would be
important for dentists to adequately screen craniofacial bony and soft tissues as well
as other general indicators such as body weight, presence of developmental changes,
and syndromic features. Sometimes, together with a cautious examination of the
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medical and dental status, radiographs (panoramic and lateral) or tomographic
images should strengthen diagnostic hypotheses and diagnostic accuracy.
302 C. D’Elia et al.
Craniofacial characteristics can be valuated both in terms
of clinical aspects and
radiographic findings using cephalometry. Classically, increased height of the face is
common in children with OSA as well as a reduction in maxillary width and retruded
mandibular prominence. Other aspects have been supported by the evidence as
characterizing pediatric OSA: a lower position of the hyoid bone, a reduction in
SNB angle, supporting the retruded mandible, and a reduced Pg-NB as indicative of
chin retrusion. Also, an initial screening for tonsils enlargement and adenoid hyper-
trophy as documented by imaging examination seems to be important in diagnosis
and referral (Lee et al.
2020).
In patients with syndromes affecting the craniofacial area, these standards often
have some distinctive markers which the dentist should be aware of. Orthodontic and
orthopedic strategies can be put forward to achieve treatment success. Rapid max-
illary expansion is indicated for taking care of transverse maxillary constriction or
posterior cross bite and it can be used as a single option or as an adjunctive measure.
Such effects are often potentiated by a previous adenotonsillectomy as well as the
adequacy of healthy lifestyles.
17.19 Final Considerations
Pediatric OSA is a common and impacting condition with the potential to affect
more than momentum in the children’s life and have substantial influences on
familial and social status of the patient. Health-related consequences require imper-
ative and adequate managing to succeed. Screening solutions, diagnostic models,
and therapeutic standards are challenges and a multi-professional approach should
be required to successfully respond to the patient itself but also to the social and
environmental determinants.
Future improvements on each of such dimensions will certainly confirm the
advantage of a h ealthy, good sleeper human being.
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