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English translation from: Orthod Fr 2019;90:343-370
© SFODF, 2019
https://doi.org/10.1051/orthodfr/2019035 Original article
The contribution of orofacial myofunctional reeducation
to the treatment of obstructive sleep apnoea
syndrome (OSA): a systematic review of the literature
Philippe AMAT1, Éric TRAN LU Y2
1 19 place des Comtes du Maine, 72000 Le Mans, France
2 5 avenue de la Convention, 94110 Arcueil, France
Correspondance: amatphilippe@outlook.com
ABSTRACT – Introduction: Obstructive sleep apnoea syndrome (OSA) is a wides-
pread and under-diagnosed condition, making it a major public health and safety pro-
blem. Orofacial myofunctional reeducation (OMR) has been shown to be eective in the
multidisciplinary treatment of OSA in children, adolescents and adults and is prescribed
at several stages of OSA management. Objectives: e main objective of this systematic
literature review was to evaluate the eectiveness of active or passive orofacial myo-
functional reeducation (OMR) in the treatment of obstructive sleep apnoea syndrome
in children, adolescents and adults. Methods: e systematic literature review was un-
dertaken from the three electronic databases: Medline (via PubMed), Cochrane Library,
Web of Science Core Collection, and supplemented by a limited grey literature search
(Google Scholar) in order to identify the studies evaluating the eectiveness of the OMR
on OSA. e primary outcome of interest was a decrease in the Apnea–Hypopnea Index
(AHI) of at least ve episodes per hour compared to the baseline state. Secondary outco-
mes were an improvement in subjective sleep quality, sleep quality measured by night
polysomnography and subjectively measured quality of life. Results: Only ten studies
met all the inclusion criteria. Eight were randomized controlled clinical trials, one was
a prospective cohort study and another was a retrospective cohort study. Six studies were
devoted to adult OSA and four to pediatric OSA. All included studies were assessed as
“low risk of bias” based on the 12 bias risk criteria of the Cochrane Back Review Group.
Based on the available evidence, RMO allows a signicant reduction in AHI, up to
90.6% in children and up to 92.06% in adults. It signicantly reduces the intensity and
frequency of snoring, helps reduce daytime sleepiness, limits the recurrence of OSA symp-
toms after adenoamygdalectomy in children and improves adherence to PPC therapy.
Passive RMO, with the assistance provided to the patient by wearing a custom orthosis,
increases adherence to reeducation, signicantly improves snoring intensity, AHI and
signicantly increases the upper airway. Conclusions: Published data show that orofa-
cial myofunctional rééducation is eective in the multidisciplinary treatment of OSA in
children, adolescents and adults and should be widely prescribed at several stages of OSA
management. Passive RMO, with the pearl mandibular advancement orthosis designed
by Michèle Hervy-Auboiron, helps to compensate for the frequent non-compliance obser-
ved during active RMO treatments.treatment.
KEYWORDS:
Sleep Disordered Breathing /
Obstructive sleep apnea
syndrome /
Orthodontics /
Orofacial myofunctional
rééducation /
Prefabricated Functional
Appliances
2English translation from: Orthod Fr 2019;90:343-370
1. Background
Obstructive sleep apnoea syndrome (OSAS)
is a widespread condition characterised by ana-
tomical and/or functional collapse of the upper
airways (UA) leading to reduced (hypopnoea) or
arrested (apnoea) airflow, oxygen desaturation
and fragmented sleep, accompanied by respiratory
effort90,106. Most patients with OSAS remain undia-
gnosed110, making OSAS a major public health and
safety issue. In children, the many clinical symptoms
of OSA have led to the individualisation of two phe-
notypes, childhood OSAS proper and adolescent
OSAS31.
Untreated childhood OSAS can lead to cognitive
impairment1,12 6 which appears to be irreversible12,
behavioural problems94,126, growth retardation14,24,
cardiovascular38 ,111 and metabolic complications51, 57 .
If left untreated, adolescent OSAS can also cause
a wide range of cognitive and behavioural pro-
blems45, from attention disorders107 to depression,
sometimes with risk-taking behaviour and suicidal
tendencies128 , cardiovascular129 and metabolic com-
plications93.
Untreated adult OSAS is associated with an in-
creased risk of health problems, including cardio-
vascular disease49,80,99, carbohydrate-lipid metabo-
lism disorders16,40 and cancers86,91,109. Drowsiness
and impaired alertness associated with untreated
OSAS have also been shown to increase the risk of
work-related injuries and road traffic accidents47,1 0 5 ,
in relation to the fragmentation of sleep induced by
abnormal breathing events.
Orofacial myofunctional reeducation (OMR) has
been shown to be effective in the multidisciplina-
ry treatment of OSAS in children, adolescents and
adults and is prescribed at several stages of these
treatments.
The treatment37,64 of OSAS in children and ado-
lescents is based on a multidisciplinary assessment,
which makes it possible to define the therapeutic
management, surgical and medical, adapted to each
patient. C. Guilleminault54,64,76 and J. Talmant119 ,120
have drawn the attention of the medical world to
the imperative need for early diagnosis of OSAS in
children and adolescents in order to institute tar-
geted multifactorial treatments and prevent long-
term morbidity.
Adeno-tonsillectomy44,98 is the first-line surgical
treatment in children. In children and adolescents,
turbinoplasty139 by laser or radiofrequency may also
be indicated and more rarely septoplasty66, lingual
tonsillectomy71, craniofacial surgery87 and hypoglos-
sal nerve stimulation17.
Non-surgical treatment of children, and more
specifically adolescents, includes the treatment5
of overweight or obesity through dietary and psy-
chological management6, combined with increased
physical activity and a healthy sleep-wake rhythm.
Inflammatory and allergic pathologies are an indi-
cation for the prescription of anti-infectious and
anti-inflammatory treatments74, corticoids and
anti-leukotrienes. Maxillofacial abnormalities, fre-
quently associated with OSA, will benefit from ap-
propriate treatment39, rapid maxillary disjunction19
in the presence of transverse maxillary insufficien-
cy, oral appliances and functional orthopaedic ap-
pliances in the case of mandibular retrognathia in
a growing patient23, and active or passive orofacial
myofunctional reeducation (OMR) which accom-
panies any orthodontic treatment or management
of temporomandibular dysfunction (TMD)3. OMR
may also be prescribed independently of orthodon-
tic treatment as an additional medical treatment18,53,
especially after tonsillectomy134. The use of conti-
nuous positive airway pressure92 (CPAP) is reserved
for severe forms of OSAS.
Orofacial myofunctional reeducation (OMR) is
also prescribed for the multidisciplinary manage-
ment of OSA in adults.
In adults, the most effective and widely prescri-
bed non-surgical gold standard treatment is conti-
nuous positive airway pressure (CPAP). In cases of
poor compliance or intolerance to CPAP136, the oral
mandibular advancement device (OMAD) is an al-
ternative whose efficacy is comparable to that of
CPAP and is attributed to a higher adherence to
OMAD than to CPAP108 . OMAD is also indicated
as first-line therapy for moderate OSAS without
associated severe cardiovascular comorbidities58. If
necessary, a behavioural approach is proposed with
the introduction of a suitable diet, a physical activity
programme22, reduction of sleeping pills, alcoho-
lic beverages and tobacco consumption12 2, and the
use of an anti-decubitus device in case of positional
OSA89. Myofunctional reeducation (MFR), either
active or passive63, is also prescribed and has been
evaluated18,73. It contributes to improved quality of
life35, reduced snoring65 and adherence to CPAP36.
Only one recent pharmacological treatment ap-
pears to have been shown to be effective81,121.
3Amat P., Tran Lu Y E.
The surgical treatment of adult OSAS112 invol-
ves a number of proposals, including soft tissue vo-
lume reduction by tonsillectomy, which is effective
in carefully selected patients21. Nasal patency can
be optimised by nasal valve surgery, septoplasty
and turbinoplasty139 . Hypoglossal nerve stimulation
surgery is aimed at contraction of the genioglossus
muscle, the main dilator muscle of the pharynx, to
suppress pharyngeal collapse during sleep30. The hi-
ghest success rate remains that obtained by maxil-
lomandibular advancement surgery, the only real
curative treatment68.
As the dilator muscles of the pharynx play a key
role in maintaining upper airway patency during
sleep, specific muscle training methods18 and exer-
cises using various musical instruments135 , including
the didgeridoo97, have been proposed to treat OSAS.
Following Blandin’s13 recognition in 1836 of the
role played by muscle pressures on the shape of
the dental arches and his statement of the concept
of muscle balance, Rogers102 emphasised the im-
portance of functional balance, including proper
tongue placement, and the therapeutic contribution
of OMR to achieving the goals of optimal mandibu-
lar growth, facial appearance and nasal ventilation.
Since then, there have been countless treatment
proposals95 targeting the structures of the face, oral
cavity and oropharynx8,25,26,42,48,52,55,70,100,103,115,118,127,137.
OMR procedures include isotonic and isometric
exercises targeting the oral (lips, tongue) and oro-
pharyngeal (soft palate, lateral pharyngeal walls)
structures combined with specific nasal inspiration/
oral exhalation, swallowing and chewing exercises.
Their most complete description was published by
Guimaraes, et al.56.
Numerous studies have shown the effectiveness of
orofacial myofunctional reeducation (OMR) in redu-
cing the severity of OSA and associated symptoms in
adults56. Studies have also shown its effectiveness in
reducing snoring65, improving quality of life35, impro-
ving adherence to CPAP36, and treating residual OSA
after adenotonsillectomy in children53,134.
Active OMR requires significant cooperation
from the patient and, for children, constant invol-
vement of the family in the procedure. To overco-
me the frequent lack of compliance, authors have
proposed the use of passive OMR assisted by a
custom-made device29,63 or prefabricated functio-
nal appliances77,78,101,130,138,140, with for the latter the
additional objective of modifying the shape and
relationship of the dental arches. The inclusion of
these appliances in OMR procedures requires the
practitioner to undertake rigorous monitoring to
continuously assess the potential adverse effects of
the appliances on the dental arches, particularly the
vestibuloversion of the mandibular incisors.
The number of studies evaluating the effects of
OMR in OSA patients is increasing35,53,56,114,117 and
narrative reviews83,113, systematic reviews34,72,73,88,131
and meta-analyses2,18,20 are regularly published.
2. Objectives
The main objective of this systematic review of
the literature was to evaluate the effectiveness of
active and passive orofacial myofunctional ree-
ducation (OMR) for the treatment of obstructive
sleep apnoea syndrome in children, adolescents
and adults. The difference between this systematic
review and previous ones2,18,20,34,72,73,83,88,113,131 is its
global approach to orofacial myofunctional reedu-
cation (OMR), both active and passive, and its focus
on the therapeutic strategies used by the various
authors.
3. Materials and methods
The PRISMA84 statement checklist (preferred
evidence for systematic reviews and meta-analyses)
was used as a guideline for conducting this syste-
matic review. Figure 1 presents a flow chart of the
search and selection process used.
3.1. The selection criteria
The following selection criteria were used.
3.1.1. Inclusion criteria
• Population of interest
The systematic review was limited to participants
who met the following criteria: (1) polysomno-
graphic diagnosis of OSA, (2) clinical symptoms of
OSA, and (3) no syndromes and no other comorbid
conditions such as, for example, head or neck injury,
stroke, cancer or neurological disease.
• Types of studies and types of interventions
Only human clinical studies, written in English or
French, published in peer-reviewed journals, which
analysed the effects of active or passive orofacial
myofunctional reeducation (or oropharyngeal exer-
4English translation from: Orthod Fr 2019;90:343-370
cises), used alone or in combination with another
treatment such as CPAP, in patients with OSA were
included.
Studies were required to provide polysomnogra-
phic data, at least for apnoea and hypopnoea index
(AHI), before and after treatment with OMR.
Randomised clinical trials comparing OMR with
a placebo intervention or a controlled intervention
and prospective cohort studies were included. Due
to the paucity of relevant publications, retrospective
cohort studies were also sought.
3.1.2. Judging criteria
The primary endpoint of this systematic review
was a decrease in the apnoea/hypopnoea index
(AHI) of at least five episodes per hour from baseline.
Secondary endpoints were an improvement in
subjective sleep quality, sleep quality measured by
nocturnal polysomnography and subjectively mea-
sured quality of life. The benefits and possible ad-
verse effects of the strategies used and how they
may affect the functionality of the upper airways
were also recorded.
3.2. Information sources, search strategies and
identification of studies
A systematic review of the literature was under-
taken using the three electronic databases Medline
(via PubMed), Cochrane Library, Web of Science Core
Collection, in order to identify studies evaluating the
efficacy of active or passive OMR in the treatment
of OSAS in children, adolescents and adults publi-
shed up to 4 September 2019.
The search strategy and identification of relevant
studies used for the electronic search was based on
the combination of the keywords "OSA" OR "obs-
tructive sleep apnea" OR "sleep" OR "sleep apnea
syndrome" AND "myofascial rééducation" OR
"myofunctional therapy" OR "orofacial myothe-
rapy" OR "oral myotherapy" OR "tongue exercises"
OR "oropharyngeal exer- cises" OR "speech the-
rapy" OR "upper airway exercises" OR "breathing
exercises" OR "upper airway remodeling".
The eligibility of articles was determined in two
phases. In the first phase, the two authors inde-
pendently searched the three electronic databases
and screened for studies that evaluated the effec-
tiveness of OMR in the treatment of OSAS in child-
ren, adolescents and adults. If study abstracts were
not available or were not self-explanatory, the full
texts were retrieved and reviewed before a final de-
cision was made. Once potentially eligible studies
were selected, the full papers were obtained for the
second phase of the selection process.
In this second stage, the same reviewers inde-
pendently assessed the selected studies.
Articles present in several databases were only
considered once.
Both authors searched the bibliographies of the
selected articles for other relevant articles and grey
literature was partially covered by consulting Google
Scholar.
Papers that did not meet all the eligibility cri-
teria were excluded. Disagreements between the
two reviewers were discussed until a consensus was
reached.
Where data were missing, additional information
was requested from the authors of the studies by email.
3.3. Data collection
Data collection was done by both reviewers on
standardised tables, and they then reviewed the ex-
tracted information.
The authors collected the following data: name
of authors, year of publication, type of study, risk of
study bias, number of subjects, demographic cha-
racteristics (gender, age), severity of OSAS, metho-
dological parameters of OMR treatments and des-
cription of outcomes after OMR.
The data collected on the methodological pa-
rameters of OMR treatments were: therapeutic
parameters (procedures, appointment frequency,
treatment duration and therapeutic follow-up), as-
sessment and reassessment parameters, physiologi-
cal parameters (anthropometric measurements such
as body mass index (BMI) and neck circumference
before and after OMR, polysomnographic data such
as apnoea-hypopnoea index (AHI) and minimum
oxygen saturation (SpO2) before and after OMR),
symptomatology (quality of life and sleep quality
data, daytime sleepiness (Epworth Sleepiness Scale)69
and snoring intensity and frequency).
Any inaccuracies or disagreements were resolved
by re-examining the original document. If necessary,
the authors of the selected studies were contacted
and questioned about missing, inaccurate or incom-
plete data.
5Amat P., Tran Lu Y E.
3.4. Risk of bias in individual studies
Both authors independently assessed the in-
cluded studies using the twelve Cochrane Back Re-
view Group (CBRG)46 criteria for systematic reviews.
These criteria, presented in Table 1 are combined
with instructions46 adapted from Van Tulder132 , Bou-
tron, et al.15 and the Cochrane Handbook of Reviews of
Interventions60.
Each criterion should be given one of three ra-
tings: "yes", "no" or "uncertain".
A "yes" rating indicates that the criterion has
been met and therefore suggests a low risk of bias.
Studies are assessed as being at "low risk of bias"
when at least 6 of the 12 Cochrane Back Review Group
(CBRG)46 criteria have been met and the study does
not have significant biases, such as a rate of lost to
follow-up of more than 20% in one of the study
groups. Studies with serious flaws, or those in which
fewer than six of the twelve criteria are met, are as-
sessed as having a "high risk of bias".
Any discrepancies that arose during the assess-
ment of the quality of the studies were resolved by
discussion between the reviewers.
4. Results
4.1. Selection of studies
A total of 1242 articles (Medline: 238; Web of
Science Core Collection: 86; Cochrane Library: 50; Goo-
gle Scholar: 868) were initially identified with elec-
tronic searches. After analysing the available titles
and abstracts and removing duplicates, 23 articles
were selected.
One article was selected after reviewing the bi-
bliographies. A total of 24 articles were considered
eligible and warranted full reading.
After reading the 24 articles in full, 14 were ex-
cluded on the basis of the selection criteria: one stu-
dy of two cases11 and 13 studies without a control gr
oup10,27,29,59,76,77,78,82,104,116,117,133,142.
In the end, 10 articles28,35,36,53,56,63,65,85,125,134 meeting
all the inclusion criteria were selected for this syste-
matic review. The different stages of the selection
process are described in the flow diagram (Fig. 1).
4.2. Description of the included studies
Six of the ten included studies focused on adult
OSAS (Diaféria, et al.35,36, Guimaraes, et al.56, Ieto, et
al.65, Neumannova, et al.85, Torres-Castro, et al.125)
and four on paediatric OSAS (Chuang, et al.28, Guil-
leminault, et al.53, Huang, et al.63, Villa, et al.134 ).
Table 1. Sources of risk of bias46.
A 1. Was the method of randomisation adequate? Yes/No/Uncertain
B 2. Has the treatment allowance been concealed? Yes/No/Uncertain
C Has knowledge of the interventions allocated been sufficiently avoided during the study?
3. Were the patients blind to the intervention? Yes/No/Uncertain
4. Were the carers blind to the intervention? Yes/No/Uncertain
5. Was the outcome assessor blind to the intervention? Yes/No/Uncertain
D Have incomplete outcome data been adequately addressed?
6. Was the lost to follow-up rate described and acceptable? Yes/No/Uncertain
7. Were all randomised participants analysed in the group to which they were assigned? Yes/No/Uncertain
E 8. Were all results for the pre-determined outcome measures reported? Yes/No/Uncertain
F Other potential sources of bias:
9. Were the groups similar at baseline in terms of key prognostic factors? Yes/No/Uncertain
10. Were co-interventions avoided or similar? Yes/No/Uncertain
11. Was compliance acceptable in all groups? Yes/No/Uncertain
12. Was the timing of the outcome assessment the same in all groups? Yes/No/Uncertain
6English translation from: Orthod Fr 2019;90:343-370
The description of the studies, including the main
characteristics of the sample, study objectives, type
of sleep-disordered breathing, level of severity of
OSA, intervention parameters and outcome mea-
sures, is presented in Table 2.
Of the ten studies28,35,36,53,56,63,65,85,125,134 included
in this systematic review, eight were randomised
controlled trials35,36,56,63,65,85,125,134, one was a prospec-
tive cohort study28 and one was a retrospective co-
hort study53.
Two randomised controlled trials (RCTs)63,134 and
a retrospective cohort study53 have investigated the
effectiveness of OMR as a means of reducing resi-
dual OSA in children after adenotonsillectomy.
The two RCTs by Diaferia, et al.35,36 originally cor-
respond to the same study and, although they each
provide additional information, the data come from
the same subjects, which explains the similarity of
the results presented for these two studies in Re-
sults Tables 5 and 6.
All eight randomised controlled trials35,36,56,
63,65,85,125,134 included a control group, as expected for
this type of study.
In seven of these RCTs, patients in the control
group received sham OMR to minimise perfor-
mance bias, with nasal lavage and deep breathing
exercises56, head movements without therapeutic
function35,36, nasal lavage alone134, nasal lavage com-
bined with deep breathing exercises and nocturnal
nasal dilator strips during sleep65, daily one-hour
walk85, daily half-hour walk combined with dietary
and sleep recommendations125.
For the RCT by Huang, et al.63, the objective of the
study was to compare the effects of active and pas-
sive OMR with the Michèle Hervy-Auboiron man-
dibular advancement device with tongue bead28,63,79
set at 50% of maximum propulsion, and the RCT
included two groups (active and passive OMR).
Two RC Ts35,36 that compared the effects of OMR
alone or combined with CPAP included four groups
of patients (OMR, CPAP, OMR + CPAP, control).
Another RCT85 also analysed the combination of
OMR + CPAP with two groups (OMR + CPAP,
control with CPAP alone).
Patients in the control group of the prospec-
tive cohort study28 did not receive any treatment.
Figure 1
Flow diagram.
7Amat P., Tran Lu Y E.
Studies Type
of study
Characteristics
of the sample Objectives
Type and
severity of
OSAS
OMR treatment procedure
(duration, frequency, exercises) Judging criteria
Guimaraes,
et al. 200956
RCT Experimental group
N: 16
Age: 51.5±6.8
Male: 63%
AHI: 22.4±4.8
BMI: 29.6±3.8
Control group
N: 15
Age: 47.7±9.8
Male: 73%
AHI: 22.4±5.4
BMI: 31.0±2.8
To assess, in patients
with moderate OSAS, the
effects of oropharyngeal
exercises on the
objective measurement
of OSAS severity with
polysomnography as well
as on subjective sleep
symptoms including snoring,
daytime sleepiness and sleep
quality
Moderate
OSAS
Three months for all groups,
with weekly visits
Experimental group
Nasal washing and oropharyngeal
exercises
Exercises of the soft palate, tongue
and facial muscles
Exercises in ventilation, phonation,
swallowing and alternate chewing
1 x 30 min supervised session/week
1 session of 30 minutes at home/day
Control group
Nasal cleansing 3 times a day
Deep breathing exercises
through the nose in a sitting position
1 session of 30 mn supervised/week
1 x 30 min. session at home/day
Main criterion
PSG
AHI
Secondary criteria
Minimum arterial
oxygen saturation
(SaO2)
Berlin questionnaire
(snoring frequency)
Epworth sleepiness
scale [ESS]
(subjective daytime
sleepiness)
Pittsburgh Sleep
Quality Index
Diaféria,
et al. 201335
RCT Speech Therapy Only Group
N: 27
Age: 45.2±13.0
Male: 100%
AHI: 28.0±22.7
BMI: 25.0±7.4
ESS: 13.7±3,2
To evaluate the effect of
speech therapy, alone or
combined with CPAP, on the
quality of life of patients with
OSAS
Mild,
moderate
and severe
OSA
Three months for all groups
Speech Therapy Only Group
Oropharyngeal exercises
3 sessions of 20 mn at home/day
CPAP Group
Device with nasal mask, without
humidifier and set to the optimal
pressure according to the PSG of
each patient
All outcomes were
measured before
and after treatment
and after a 3-month
washout period
Table 2. Description of study data on subject characteristics, type of disorder, degree of OSA severity and treatment parameters.
.../...
8English translation from: Orthod Fr 2019;90:343-370
Studies Type
of study
Characteristics
of the sample Objectives
Type and
severity of
OSAS
OMR treatment procedure
(duration, frequency, exercises) Judging criteria
CPAP Group
N: 27
Age: 46.4±9.1
Male: 100%
AHI: 34.4±22.4
BMI: 28.7±3.3
ESS: 12.0±2.1
Speech Therapy and CPAP
Group
N: 22
Age: 47.5±10.9
Male: 100%
AHI: 30.4±19.8
BMI: 27.9±2.4
ESS: 12.0±2.6
Control group
N: 24
Age: 42.9±10.5
Male: 100%
AHI: 27.8±20.3
BMI: 28.6±4.0
ESS: 12.8±3.1
Speech Therapy and CPAP Group
Combining the two protocols
Control group
Head movements without therapeutic
function
-3 sessions of 20 mn at home/day
PSG
AHI
Minimum arterial
oxygen saturation
(SaO2)
Epworth sleepiness
scale [ESS] Functional
Outcome in Sleep
questionnaire (FOSQ)
WHOQOL-Bref
questionnaire
SF-36 Health
questionnaire
Guilleminault,
et al. 201353
Retrospec-
tive cohort
Group with myofacial
reeducation
N: 11
Age: 7.3±1.5
Boys/Girls: 23/1
AHI: 0.4±0.3
Minimum SaO2 (%): 95±1
Flow limitation
(% total sleep time): 10±10
To evaluate the impact of
myofunctional rééducation
(MR) in children with sleep
disordered breathing referred
for adenotonsillectomy,
orthodontics, and
myofunctional treatment in
three different geographical
areas
Mild,
moderate
and severe
OSA
Group with myofacial reeducation
Duration of 24 months
Tongue and orofacial muscle
strengthening exercises
Regular supervised sessions as
needed
Several sessions at home/day
PSG at diagnosis,
following
adenotonsillectomy
+ orthodontics, and at
long-term follow-up
.../...
9Amat P., Tran Lu Y E.
Studies Type
of study
Characteristics
of the sample Objectives
Type and
severity of
OSAS
OMR treatment procedure
(duration, frequency, exercises) Judging criteria
Control group
N: 13
Age: 7.3±1.5
Boys/Girls: 23/1
AHI: 0.4±0.3
Minimum SaO2 (%): 95±1
Flow limitation
(% total sleep time): 10±10
Control group
Lack of myofacial reeducation due to
non-adherence to recommendations
or abandonment
AHI
Minimum SaO2
Flow limitation
Myofunctional
assessment
Modified Mallampati
score
Villa,
et al. 2015134
RCT Experimental group
N: 14
Age: 6.01±1.55
AHI: 4.87±2.96
BMI (percentile):
81.85±29.94
Control group
N: 13
Age: 5.76±0.82
AHI: 4.56±3.22
BMI (percentile):
68.22±28.68
To evaluate the effectiveness
of oropharyngeal exercises
as a means of reducing
residual OSA in children after
adenoamygdalactomy (AA)
Residual
OSAS after
AA
Two months, three meetings
with the therapist
Experimental group
Nasal cleansing and oropharyngeal
exercises
3 sessions at home/day, with 10-20
repetitions each time
Control group
Nasal cleansing
2 sessions at home/day, morning
and evening
PSG before AA,
6 months after AA
and after 2 months of
exercise
Improvement in OSA
was defined as Δ AHI:
(AHI at T1-AHI at T2)/
AHI at T1×100
Sleep Clinical Record
(SCR) questionnaire
Morphofunctional
assessment
leto,
et al. 201565
RCT Experimental group
N: 19
Age: 48±14
Male: 57.9%
AHI: 15.6±9.3
BMI: 28.3±2.7
To assess the effects of
oropharyngeal exercises
on snoring in patients with
minimal snoring symptoms
and a diagnosis of primary
snoring or mild to moderate
OSA
Mild and
moderate
OSA
Three months for all groups,
with weekly visits
Experimental group
Nasal cleansing
Oropharyngeal exercises
1 supervised session/week
3 sessions of 8 mn at home/day
Objective snoring
index and total
snoring index
obtained after
recording snoring
during PSG
.../...
10 English translation from: Orthod Fr 2019;90:343-370
Studies Type
of study
Characteristics
of the sample Objectives
Type and
severity of
OSAS
OMR treatment procedure
(duration, frequency, exercises) Judging criteria
Control group
N: 20
Age: 45±13
Male: 55%
AHI: 15.1±9.5
BMI: 28.3±2.5
Control group
Nasal cleansing
Deep breathing exercises
Nasal dilation strips during sleep
1 supervised session/week
3 sessions of 8 mn at home/day
AHI
Epworth sleepiness
scale [ESS]
(subjective daytime
sleepiness)
Pittsburgh Sleep
Quality Index
Anthropometric
assessment
Diaféria,
et al. 201736
RCT Myofunctional Therapy
(MFT) group alone
N: 27
Age: 45.2±13.0
Male: 100%
AHI: 28.0±22.7
BMI: 25.0±7.4
ESS: 13.7±3.2
CPAP Group
N: 27
Age: 46.4±9.1
Male: 100%
AHI: 34.4±22.4
BMI: 28.7±3.3
ESS: 12.0±2.1
MFT + CPAP Group
N: 22
Age: 47.5±10.9
Male: 100%
AHI: 30.4±19.8
BMI: 27.9±2.4
ESS: 12.0±2.6
Control group
N: 24
Age: 42.9±10.5
Male: 100%
AHI: 27.8±20.3
BMI: 28.6±4.0
ESS: 12.8±3.1
Assessing the effect of
myofunctional treatment on
CPAP compliance
Mild,
moderate
and severe
OSA
Three months for all groups, with
weekly visits for the placebo, MFT
and MFT + CPAP groups
Three visits for the CPAP group
MFT Group
Oropharyngeal exercises
3 sessions of 20 mn at home/day
CPAP Group
Device with nasal mask, without
humidifier and set to the optimal
pressure according to the PSG of
each patient
MFT + CPAP Group
Combining the two protocols
Control Group
Head movements without therapeutic
function
3 sessions of 20 mn at home/day
All outcomes were
measured before
and after treatment
and after a 3-week
washout period
Assessment of
compliance with
CPAP
PSG
AHI
Epworth sleepiness
scale [ESS] Subjective
assessment of
snoring intensity and
frequency
Myofunctional
assessment
Modified Mallampati
score
.../...
11Amat P., Tran Lu Y E.
Studies Type
of study
Characteristics
of the sample Objectives
Type and
severity of
OSAS
OMR treatment procedure
(duration, frequency, exercises) Judging criteria
Huang,
et al. 201863
RCT A-MFT Group
(Active MFT)
N: 54
Age: 7.02±2.44
Boys: 50%
BMI: 15.61±1.74
AHI: 2.47±1.31
P-MFT Group
(Passive MFT)
N: 56
Age: 7.97±3.08
Boys: 64.6%
BMI: 17.04±3.05
AHI: 6.00±7.23
To evaluate the impact of
active (A-MFT) or passive
(P-MFT) myofunctional
therapy in children
with residual OSA after
adenoamygdalectomy (AA)
and children with OSA
without indication for AA
Moderate
and severe
OSAS
12 months for all groups
A-MFT Group (Active MFT)
Isometric and isotonic exercises of
the soft palate, tongue and facial
muscles
Exercises in ventilation, phonation,
swallowing and alternate mastication
1 session of 20 mn at home/day
Regular supervised sessions
P-MFT Group (Passive MFT)
Nightly wearing of the mandibular
advancement device with tongue
bead set at 50% of maximum
mandibular propulsion
Monthly check-up by the orthodontist
Clinical and PSG
assessment at
baseline, 3 months,
6 months and
12 months
AHI
Cephalometric
assessment at
baseline, 6 and
12 months
Neumannova,
et al. 201885
RCT Experimental group
(CPAP +PR)
N: 15
Age: 53.87±7.79
Neck circumference:
44.9±10.3
BMI: 40.3±9.4
AHI: 54.2±27.4
Control group (CPAP)
N: 20
Age: 54.05±5.53
Neck circumference:
43.5±4.6
BMI: 36.4±5.4
AHI: 55.4±28.9
To compare the short-term
effects of CPAP combined
with a reeducation
programme, combining
pulmonary rehabilitation,
oropharyngeal and facial
exercises, with CPAP, on
OSA severity, ventilation
and changes in body
characteristics of a newly
diagnosed patient.
Moderate
and severe
OSAS
6 weeks for all groups
Experimental group (CPAP +PR)
Individual aerobic training combined
with therapeutic education, breathing
exercises and respiratory muscle
training
2 sessions of 60 mn/week with a
personal physiotherapist
Oropharyngeal exercises: soft palate,
tongue and facial muscle exercises
Control group (CPAP)
A minimum of 60 mn of walking per
day
All outcomes were
measured before and
after treatment
Main criterion
PSG
AHI
Oxygen Desaturation
Index
Epworth Sleepiness
Scale
Mean nocturnal SpO2
Secondary criteria
Percentage of total
sleep time with
oxygen saturation
below 90%.
BMI
Vital capacity and
forced expiratory
volume in 1 s
Neck, waist and hip
circumference
.../...
12 English translation from: Orthod Fr 2019;90:343-370
Studies Type
of study
Characteristics
of the sample Objectives
Type and
severity of
OSAS
OMR treatment procedure
(duration, frequency, exercises) Judging criteria
Torres-Castro,
et al. 2019125
RCT Experimental group
N: 14
Age: 64.5 (51.8–74)
Male: 53.8%
AHI: 30.5 (22.5–41.3)
BMI: 31.3 (27.5–35)
Neck circumference: 40
(37–41.5)
Control group
N: 13
Age: 67 (53–74.5)
Male: 57.1%
AHI: 37 (25.5–43.5)
BMI: 27.1 (25.1–35.9)
Neck circumference: 38
(37–41.5)
To evaluate the effects of
a combined physical and
oropharyngeal exercise
programme on the apnoea-
hypopnoea index in patients
with moderate to severe
Moderate
and severe
OSAS
8 weeks for all groups
Experimental group
Brisk walking in urban areas for
30 mn, 3 times a week, usually
supervised by a physiotherapist
Oropharyngeal exercises
Nutrition and sleep
recommendations
Control group
Recommendations for diet and sleep
Brisk walk for 30 minutes, at least 3
times a week
All outcomes were
measured before and
after treatment
PSG or NRP
AHI
Oxygen Desaturation
Index
Epworth Sleepiness
Scale
Quebec Sleep
Questionnaire
Hospital Anxiety and
Depression Scale
(HADS)
BMI
Neck, waist and hip
circumference
Chuang,
et al. 201928
Prospective
cohort
P-MFT Group
(Passive MFT)
N: 17
Age: 7.95±3.27
Boys: 77.5%
BMI: 17.60±3.72
AHI: 3.09±2.56
Control group
N: 40
Age: 7.65±2.70
Boys: 76.5%
BMI: 19.25±3.98
AHI: 3.09±2.56
To evaluate the effects
of one year of passive
myofunctional therapy
(P-MFT) on craniofacial and
airway morphology and
quality of life in children with
OSA
Moderate
OSAS
12 months for all groups
Experimental group (Passive MFT)
Nightly wearing of the mandibular
advancement device with tongue
bead set at 50% of maximum
mandibular propulsion
Checked every 3 months by the
orthodontist
Control group
No treatment
All outcomes were
measured before and
after treatment
PSG
AHI
Lateral cephalometric
assessment
Quality of life
questionnaire
(OSA-18)
Abbreviations: RCT, randomised controlled trial; OSAS, obstructive sleep apnoea and hypopnoea syndrome; BMI, body mass index; PSG, polysomnography;
NRP, nocturnal respiratory polygraphy; AHI, apnoea-hypopnoea index; CPAP, continuous positive airway pressure; PR, pulmonary rehabilitation, oropharyngeal
and facial exercises program.
13Amat P., Tran Lu Y E.
The control group in the retrospective cohort stu-
dy53 consisted of patients who did not complete the
myofacial reeducation program due to non-com-
pliance or drop-out.
Active, non-device based OMR procedures diffe-
red between studies in the choice of exercises, num-
ber of repetitions, frequency and duration of daily
practice (from six weeks to one year), while main-
taining a common approach. The authors mainly
used a series of "oropharyngeal" exercises, derived
from speech therapy and physical therapy. Isometric
and isotonic exercises were performed to optimise
muscle tone and mobility, to adjust the position of
the soft tissues (soft palate, pharyngeal constric-
tor muscles, supra-hyoid muscles, tongue, cheeks
and lips) and to improve the orofacial functions of
ventilation, mastication, swallowing and phonation.
The authors did not provide any justification for
their choice of the type of exercises, frequency and
duration of rehabilitation sessions. Table 3 shows
an example of oropharyngeal exercises used in the
Neumannova RCT85.
The potential for the effects of OMR to persist
remains an issue, particularly for orthodontists. Take
the example of lip tone exercises, which are often
prescribed to help the patient regain lip compe-
tence. One study showed that lip training with an
oral screen for nine months did increase lip strength,
but that it then decreased as measured ten months
after the muscle training stopped123. It may therefore
seem appropriate to extend the OMR therapy pro-
grams to maintain the results obtained previously
Type of exercises Description of the exercises
Tongue exercises - Push anterior half of the tongue against the hard palate for 5 s., keep the jaw open
throughout the exercise, relax the tongue for 8 s., 10 repetitions, three times a day.
- Open the mouth widely, try to touch the chin with the tip of the tongue, hold this position
for 5 s., place the tongue into the mouth and relax for 8 s., 10 repetitions, three times a day.
Soft palate exercises - Pronounce an oral vowel “A, E, I, O, U” intermittently (isotonic exercise) and continuously
(isometric exercise), five repetitions, once a day.
- Breathe in through the nose, breathe out through the mouth, during breathe out period
press the lips together, maintain the blowing for 5 s., five repetitions, three times a day.
Exercises for the cheeks,
throat, and neck
- Tilt the head back, stick the tongue out and upward (“try to touch the ceiling with the tip
of the tongue”), hold it for 5 s., then move the head into an upright position and relax the
tongue in the mouth for 8 s., 10 repetitions, once a day.
- Tilt the head back, gently bite the tongue and try to swallow once, then move the head in
an upright position and relax the tongue in the mouth for 8 s., five repetitions, once a day.
- Place the index finger inside the cheek, place the thumb on outside the cheek, pull the
cheek outward with the fingers, at the same time contract the cheek muscle to resist the
pulling for 5 s., relax for 8 s., 10 repetitions, once a day.
Exercises for lips and jaw - Purse the lips, hold the position for 10 s., relax for 12 s., five repetitions, once a day.
- Purse the lips with the mouth wide open, hold the position for 5 s., relax for 8 s., five
repetitions, once a day.
- Place a hand under the chin, attempt to open the mouth for 5 s., but with the hand pushing
against the lower jaw (the task is to stop the mouth opening), relax for 8 s., 10repetitions,
once a day.
* Oral vowels are pronounced with the soft palate raised, which closes off the passage of air through the nose. Nasal vowels are
pronounced with the soft palate lowered, allowing air to pass through the mouth and nose.
Table 3. Description of oropharyngeal exercises used in the Neumannova RCT85.
14 English translation from: Orthod Fr 2019;90:343-370
and to combine therapeutic education with OMR3.
This issue is even more important for OMR treat-
ments for OSAS, which is a particularly restrictive
approach and requires unfailing patient compliance,
which explains the many dropouts. The only studies
that have raised this important issue of compliance
are a retrospective cohort study53, with assess-
ment between 22 and 50 months after completion
of OMR, and four RCTs28,35,36,63. Two of the RCTs
addressed this issue with medium- and long-term
follow-up28,63 and two repeated the measures after
a 3-week washout period35,36.
The data collected in the ten studies in this syste-
matic review were physiological or symptomatological.
The main physiological data were either obtained
by polysomnography, for apnoea and hypopnoea
index per hour of sleep (AHI) and minimum blood
oxygen saturation (minimum SpO2), or were an-
thropometric measurements, including body mass
index (BMI) and neck circumference.
The main symptomatological data included mea-
sures of quality of life, including the Whoqol-Bref,
Functional Outcome in Sleep Questionnaire (FOSQ) and
the SF-36 health questionnaire, and of sleep quality,
using the Pittsburgh Sleep Quality Index. Subjective
daytime sleepiness with the Epworth Sleepiness
Scale (ESS) and snoring intensity and frequency with
a visual analogue scale, the Berlin Questionnaire and
snoring recording during polysomnography were
also assessed.
4.3. Methodological quality of the included study
All included studies met at least six of the Co-
chrane Back Review Group (CBRG)46 criteria (Tab. 1)
and were not subject to significant bias, such as too
many patients being lost to follow-up in one of the
study groups, so they were all assessed as being at
"low risk of bias" (Tab. 4).
Type of exercises
Methodological quality according to
the CBRG46 risk of bias criteria Tot al Quality Conflit of
interest
123456789101112
Guimaraes,
et al. 200956 NS NYNYYYYYYYY 9 High No
Diaféria,
et al. 201335 NS N Y N NS N Y Y Y Y Y Y 7 High No
Guilleminault,
et al. 201353 NS N N N Y Y Y Y Y Y Y Y 8 High No
Villa,
et al. 2015134 NS N Y N Y Y Y Y Y Y Y Y 9 High No
leto,
et al. 201565 NS N Y N Y Y Y Y Y Y Y Y 9 High No
Diaféria,
et al. 201736 NS N Y N Y N Y Y Y Y Y Y 8 High No
Huang,
et al. 201863 NS N N N Y N Y Y Y Y N Y 6 High No
Neumannova,
et al. 201885 NS N Y N N Y Y Y Y Y Y Y 8 High No
Torres-Castro,
et al. 2019125 Y Y Y N Y Y Y Y Y Y Y Y 11 High No
Chuang,
et al. 201928 NS N N N Y Y Y Y Y Y Y Y 8 High No
Y: yes, N: no, NS: not specified by the authors of the study.
Table 4. Methodological quality of studies assessed according to the Cochrane Back Review Group46 risk of bias criteria.
15Amat P., Tran Lu Y E.
Studies
Anthropometric measurements Polysomnography
BMI (kg/m 2 ) NC (cm) AHI (nb/hour) SpO2 minimum (%)
Before After Before After Before After Before After
Guimaraes,
et al. 200956
29.6±3.8 29.5±4.3 39.6±3.6 38.5±4.0*
(P=0.01)
22.4±4.8 13.7±8.5*
(P<0.01)
83±6 85±7*
Diaféria,
et al. 201335 ∏
25.0±7.4 26.7±2.9 41.6±3.7 41.5±2.3 28±2.7 13.9±18.5*
(P<0.001)
83.7±7.7 84.9±8.8
Guilleminault,
et al. 201353
NR NR NR NR 5.3±0.3
(without OMR)
0.5±0.4* (with OMR)
(P=0.001)
91±1.8 96±1*
x2 test (P=0.1)
Villa,
et al. 2015134
81.85±29.94 NR NR NR 4.87±2.96 1.84*
(P=0.004)
NR NR
leto,
et al. 201565
28.1±2.7 28.2±2.8 37.9±2.5 37.5±2.4**
(P<0.05)
25.4 (22.1-28.7)
18.1 (15.4-24.1) ***
(P=0.17)
85.5±7.5 83.8±8.9
Diaféria,
et al. 201736 ∏
25.0±7.4 26.7±2.9 41.6±3.7 41.5±2.3 28±22.7 13.9±18.5*
(P<0.001)
83.7 ±7.7 84.9±8.8
Huang,
et al. 201863 Ω
17.04±3.05 18.53±3.99 NR NR 6.00±7.23 2.44±2.28*
(P=0.001)
NR NR
Neumannova,
et al. 201885
40.3±9.4 39.6±9.1*
(P<0.01)
44.9±10.3 43.7±3.5*
(P<0.005)
54.2±27.4 4.3±3.9*
(P<0.0001)
NR NR
Torres-Castro,
et al. 2019125
31.3 (27.5–35) 30.2 (27.3–34.7)*
(P=0.003)
40(37–41.5) 38.8(37.4–40.5) 30.5(22.5–41.3) 34.5(14.5–45) NR NR
Chuang,
et al. 201928
17.6±3.72 NR NR NR 3.75±2.48 2.16±1.80*
(P=0.002)
SaO2 minimum
90.13±4.01
SaO2 minimum
91.95±3.56*
(P=0.001)
Table 5. Results of orofacial myofunctional reeducation (OMR) in terms of physiological data.
Data are presented with their mean and standard deviation, except for presented with the median, minimum and maximum.
Ω: only the results of the group with passive myofunctional therapy (P-MFT) were presented, as no patients in the active myofunctional therapy (A-MFT) group completed the study.
∏: only results from the OMR group are presented.
Abbreviations: OMR, orofacial myofunctional reeducation; NR, no record of this data, which did not correspond to the objective of the study or which the study authors did not retain;
BMI, body mass index; NC, neck circumference; AHI, apnoea and hypopnoea index per hour of sleep; SpO2 minimum, minimum blood oxygen saturation; CPAP, continuous positive
airway pressure; *, P statistically significant (<0.05) T-test; **, P statistically significant for comparisons using repeated measures analysis of variance; ***, P variation only for the
moderate OSAS group.
16 English translation from: Orthod Fr 2019;90:343-370
Studies
Symptomatology
Quality of life
Quality of sleep ESS Intensity of snoring Frequency of snoring
Before After Before After Before After Before After
Guimaraes,
et al. 200956
Pittsburgh
10.2 ± 3.7
Pittsburgh
6.9 ± 2.5*
14 ± 5 8 ± 6*
(P=0.01)
3 (3-4) 1 (1-2) *
(P=0.001)
4 (4-4) 3 (1.5 - 3.5) *
(P=0.001)
Diaféria,
et al. 201335 ∏
FOSQ
WHOQOL-Bref
SF-36
FOSQ
WHOQOL-Bref*
(P<0.001)
SF-36
13.7 ± 3.2 7.5 ± 3.7*
(P<0.001)
8.5 ± 2.3 4.9 ± 3.2*
(P<0.001)
7.7 ± 2.3 4.3 ± 2.8*
(P<0.001)
Guilleminault,
et al. 201353
NR NR NR NR NR NR NR NR
Villa,
et al. 2015134
SCR
4.07 ± 2.22
NR NR NR NR NR NR NR
leto,
et al. 201565
Pittsburgh
6 ± 3.2
Pittsburgh
4 ± 2.6*
(P=0.04)
7 (3-11) 7 (4-10) 2 (2-3) S
4 (2.5-4) P
2 (1-2) S
1 (1-2) P*
(P=0.03)
3 (2-4) S
4 (3-4) P
2 (1-4) S
2 (1.5-3) P*
(P=0.04)
Diaféria,
et al. 201736 ∏
NR NR 13.7 ± 3.2 7.5 ± 3.7*
(P<0.001)
8.5 ± 2.3 4.9 ± 3.2*
(P<0.001)
7.7 ± 2.3 4.3 ± 2.8*
(P<0.001)
Huang,
et al. 201863 Ω
NR NR NR NR Snoring index
(nb/h)
212.91
Snoring index
(nb/h)
83.16* (P=0.046)
NR NR
Neumannova,
et al. 201885
NR NR 12.9 ± 4.7 5.7 ± 4.1 *
(P<0.0005)
NR NR NR NR
Torres-Castro,
et al. 2019125
QSQ
180.5(151–200)
QSQ
186(153.8–201)
8 (3–13) 8 (4–10.3) NR NR NR NR
Chuang,
et al. 201928
OSA-18
55.85 ± 17.44
OSA-18
45.51 ± 14.44*
(P=0.000)
NR NR Snoring index
(nb/h)
212.91
Snoring index
(nb/h)
212.91
NR NR
Data are presented with their mean and standard deviation, except for presented with the median, minimum and maximum.
Ω: only the results of the group with passive myofunctional therapy (P-MFT) were presented, as no patients in the active myofunctional therapy (A-MFT) group completed the study.
∏: only the results of the group with OMR alone are presented.
Abbreviations: NR, no record of this data, which did not fit the study objective or was not retained by the study authors; ESS, Epworth Sleepiness Scale (subjective daytime sleepiness);
*, P significant (<0.05) T-test; S, information given by the research subject; P, information given by the sleep partner; QSQ, Quebec Sleep Questionnaire; OSA-18, OSA-18 Quality
of Life Questionnaire.
Table 6. Results of orofacial myofunctional reeducation (OMR) in terms of symptomatological data.
17Amat P., Tran Lu Y E.
4.4. Results of the included studies
4.4.1. General presentation of the results
The main results of orofacial myofunctional reha-
bilitation (OMR) have been grouped in two tables,
one where the results are described in terms of phy-
siological data (Tab. 5), the other in terms of symp-
tomatological data (Tab. 6).
4.4.2. Results obtained with active OMR
Active OMR is defined as OMR that is not asso-
ciated with the use of a custom-made appliance29,63
or prefabricated functional appliances77,78,101,130,138,140
as is passive OMR.
The patient performs isotonic and isometric exer-
cises that target the oral, oropharyngeal structures
and are combined with specific ventilation, swal-
lowing and chewing exercises. As dysfunction of the
orofacial and pharyngeal muscles33 and impaired
oropharyngeal control can contribute to airway col-
lapse41, a contribution of active OMR to the mana-
gement of OSA is sought.
4.4.2.1. Effects of active OMR on physiological variables
4.4.2.1.1. Effects of active OMR on PSG variables
Four35,36,56,85 of the six RCTs dedicated to adults
showed a significant reduction in AHI.
The three studies in children, two RCTs63,134 and
one prospective cohort study7, all showed a signifi-
cant decrease in AHI.
A statistically significant increase in the percen-
tage of arterial minimum oxygen saturation was
found in an adult RCT56 and a prospective study in
children28.
4.4.2.1.2. Effects of active OMR on anthropometric
variables
Measurements of neck circumference, abdominal
circumference and BMI are anthropometric predic-
tors of the severity of OSAS96,124, 62. Pharyngeal cri-
tical closure pressure is associated with obesity50,
decreased upper airway elasticity and is observed in
obese patients with a large neck circumference61,6 7.
The statistically significant decrease in neck cir-
cumference measured in the studies of Guimaraes,
et al.56, Ieto, et al.65, Neumannova, et al.85 suggests
the possibility of upper airway remodelling by active
OMR exercises. The decrease in neck circumference
is correlated with a statistically significant reduction
in AHI in the studies by Guimaraes, et al.56 and Neu-
mannova, et al.85.
A statistically significant decrease in BMI is re-
ported in the only study by Neumannova, et al.85.
4.4.2.1.3. Effects of active OMR on orofacial
myofunctional status
Assessment of myofunctional status should
help to determine whether the effects of OMR in
OSAS patients are related to improved muscle and
orofacial function. Surprisingly, only the studies by
Guilleminault, et al.53, Villa, et al.134 and Diaféria, et
al.36 included a myofunctional assessment. Interpre-
tation and comparison of the results could not be
conducted due to the lack of a standardised assess-
ment tool.
In the retrospective cohort study by Guillemi-
nault, et al.53, myofunctional assessment of the oro-
facial region of the control group, who had not had
myofacial reeducation due to non-compliance or
drop-out, showed that the patients had an abnor-
mally low tongue position in the mouth when awake.
Twelve of the thirteen patients in the control group
were unable to make tongue clicks, ten were unable
to move their tongue upwards when asked to try to
touch their nose with the tip of their tongue, four
had difficulty holding a button between their lips,
and one had difficulty swallowing when drinking ra-
pidly. All subjects admitted to having a preferential
side of unilateral chewing, and nine subjects had a
slight asymmetry of the masseter muscles when as-
sessing active contraction. Finally, all subjects were
assessed with abnormal orofacial muscle tone when
awake.
In contrast, all eleven patients in the OMR group
were rated as normal on myofunctional assessment.
Diaféria, et al.36 reported that the modified Mal-
lampati score improved in the OMR and CPAP +
OMR groups and was correlated with increased
tongue and soft palate strength.
Villa, et al.134 evaluated the effectiveness of oro-
pharyngeal exercises as a means of reducing residual
OSA in children after adenotonsillectomy. The exer-
cises prescribed were of three types: (1) ventilatory
rehabilitation, (2) lip closure and lip tone exercises,
and (3) tongue posture exercises. Active OMR signi-
ficantly increased the number of patients with nasal
18 English translation from: Orthod Fr 2019;90:343-370
ventilation, effective lip closure and good lip tone.
No significant improvement was observed for pa-
tients in the control group.
4.4.2.2. Effects of active OMR on symptomatological
variables
4.4.2.2.1. Effects of active OMR on quality of life
and sleep quality
The studies by Diaféria, et al.35,36 and Chuang, et
al.28 showed a statistically significant improvement
in quality of life. The RCTs of Diaféria, et al.35,36 re-
ported this improvement in quality of life for the
OMR and OMR + CPAP groups, but not for the
CPAP and control groups.
The studies by Guimaraes, et al.56 and Ieto, et al.65
showed a statistically significant improvement in
sleep quality.
4.4.2.2.2. Effects of active OMR on daytime sleepiness
The Epworth Sleepiness Scale (ESS), proposed
by Johns69, is generally used to assess daytime slee-
piness. The ESS assesses the propensity to sleep in
eight different situations, with a total score ranging
from zero (absence) to 24 (intense).
The RCT of Guimaraes, et al.56 reported a statis-
tically significant improvement in ESS at the end of
treatment with OMR. The RCTs of Diaféria, et al.35,36
also showed a significant improvement in ESS at the
end of OMR treatment in the OMR, OMR + CPAP
and CPAP groups. Measurement of ESS at the end
of three weeks of therapeutic withdrawal after in-
tervention showed that the improvement in SSE
remained statistically significant in the OMR and
CPAP groups.
4.4.2.2.3. Effects of active OMR on snoring intensity
and frequency
Jacques Talmant, et al.12 0 pointed out that struc-
tural changes, secondary to the vibratory trauma
caused by snoring, can affect each component of
the pharyngeal structures43 and contribute to the
collapsibility of this segment of the airways. They
may increase the risk of carotid atherosclerosis75 and
bilateral carotid artery stenosis32.
A statistically significant decrease in snoring
intensity and frequency was observed in studies
conducted by Guimaraes, et al.56, Ieto, et al.65 and
Huang, et al.63.
The studies by Diaféria, et al.35,36 showed a signi-
ficant decrease in snoring intensity and frequency
for the OMR, OMR + CPAP and CPAP groups af-
ter treatment and in the OMR + CPAP and CPAP
groups when compared to the control group.
At the end of three weeks of therapeutic wit-
hdrawal after intervention, the reduction in snoring
intensity and frequency was statistically significantly
maintained in the OMR group but did not persist in
the OMR + CPAP and CPAP groups.
4.4.2.3. Contribution of active OMR to the treatment
of OSA with continuous positive airway
pressure (CPAP)
The objective of the RCT by Diaferia, et al.35
was to assess the effect of OMR, either alone or
in combination with CPAP, on the quality of life of
patients with OSAS. Analysis of the quality of life
data showed a statistically significant improvement
in the physical health dimension (Whoql-Bref ques-
tionnaire) for the OMR and OMR + CPAP groups.
The functional capacity dimension (SF-36 health
questionnaire) improved statistically significantly in
the OMR group. Quality of life, assessed with the
FOSQ, did not change significantly in any of the four
study groups.
The same team of Diaféria, et al. had evaluated
the effect of OMR on adherence to CPAP in ano-
ther RCT36. Although OMR did not reduce the le-
vel of CPAP required, it significantly increased ad-
herence to CPAP. The mean adherence was 55% in
the control group, 63% in the OMR group, 30% in
the CPAP group and 65% in the combined CPAP +
OMR group. Weekly therapy education and support
during the sessions may have contributed to grea-
ter adherence to CPAP when combined with OMR
(65%) than when prescribed alone (30%).
4.4.3. Results obtained with the passive OMR
To address the frequent lack of compliance ob-
served in OMR programs, authors have proposed
the use of passive OMR. This is named to reflect
the assistance provided to the patient by wearing a
custom-made appliance29,63 or prefabricated func-
tional appliances77,78,101,130,138,140, the latter with the
additional objective of modifying the shape and
relationship of the dental arches. The inclusion of
these appliances in the OMR procedures requires
the practitioner to follow up rigorously, in order to
continuously assess the potential adverse effects of
the appliances on the dental arches, especially the
vestibuloversion of the mandibular incisors.
19Amat P., Tran Lu Y E.
The studies by Levrini77,78 investigated the effect
of a prefabricated myofunctional device for the
treatment of mild to moderate paediatric obstruc-
tive sleep apnoea. They could not be included in
the systematic review due to insufficient methodo-
logical quality, mainly related to the absence of a
control group.
Studies by Huang, et al.63 and Chuang, et al.28
investigated the effects of passive OMR using the
custom-made one-piece mandibular advancement
orthosis designed by Michèle Hervy-Auboiron (pa-
tent number: EP 13753289.1; US14/420499). It is
attached to the maxilla by stabilizing clasps placed
between the maxillary second premolars and first
molars. It has a freely rotating ball that is set at the
anteroinferior end of the orthosis. The orthosis is
constructed with moderate mandibular propul-
sion, at 50% of maximum mandibular advance-
ment. The patient wears the orthosis only during
sleep. The patient is asked to rotate the ball just be-
fore falling asleep in order to obtain lingual propul-
sion and upper airway clearance. The objective is
to induce a persistent lingual propulsion response
in case of apnea.
The study by Huang, et al.63 aimed to assess the
impact of active or passive OMR in children with
residual OSA after adenotonsillectomy (AA) and in
children with OSA without an indication for AA. It
showed a statistically significant decrease in snoring
intensity and AHI. Cephalometric assessment of the
subjects showed no adverse effects from nighttime
use of the orthosis set at 50% of maximum mandi-
bular propulsion, and a significant increase in upper
airway.
The study by Chuang, et al.28 evaluated the effects
of one year of passive OMR on the craniofacial and
airway morphology and quality of life of children
with OSA. She reported a statistically significant im-
provement in quality of life (OSA-18) and AHI du-
ring sleep and REM AHI. Cephalometric assessment
of the patients showed significant improvement in
linear mandibular growth (Co-Gn) and upper airway
morphology.
5. Discussion
5.1. Summary of the main evidence
This systematic review, which includes ten stu-
dies on the effects of OMR on OSAS, six in adults
(Ieto, et al.65, Diaféria, et al.35,36, Torres- Castro, et
al.125 , Guimaraes, et al.56, Neumannova, et al.85) and
four in children (Chuang, et al.28, Guilleminault, et
al.53, Huang, et al.63, Villa, et al.134 ), leads to five main
conclusions.
5.1.1. OMR allows for a reduction in AHI
In adults, OMR provides a statistically signifi-
cant reduction in AHI from 28.74% (Ieto, et al.65)
to 92.06% (Neumannova, et al.85), a study that re-
ported a decrease in pre- and post-OMR AHI from
54.2 ± 27.4 to 4.3 ± 3.9* (P < 0.0001).
In children, OMR provides a statistically signifi-
cant reduction in AHI of up to 90.6% in the retros-
pective cohort study by Guilleminault, et al.53, in
which the M ± SD was 0.5 ± 0.4* (P = 0.001) with
OMR and 5.3 ± 0.3 without OMR. The RCT that re-
ported the largest significant decrease was Villa, et
al.134 , with a mean AHI that decreased by 62.22%,
from 4.87 ± 2.96 before OMR to 1.84*, P = 0.004
after OMR.
The persistence of the effect of OMR on AHI is
variable. The study by Diaféria, et al.35 shows a de-
crease in AHI significance after three months of
OMR, and a loss of significance after three weeks of
therapeutic withdrawal. The study by Guilleminault,
et al.53 reported that eleven children remained cured
of OSA (AHI of 0.5 ± 0.4/h) 22 to 50 months after
completion of the OMR program.
5.1.2. OMR allows a reduction in the intensity
and frequency of snoring
OMR can significantly reduce snoring, both sub-
jectively and objectively (Guimaraes, et al.56, Ieto, et
al.65, Huang, et al.63, Diaféria, et al.35,36). The studies
by Diaféria, et al.35,36 reported a persistence of the ef-
fect of OMR with the maintenance of the decrease
in snoring intensity and frequency in the OMR
group at the end of the three weeks of therapeutic
withdrawal after intervention.
5.1.3. OMR helps to reduce daytime sleepiness
OMR can reduce daytime sleepiness, as evidenced
by statistically significant improvement in ESS at the
end of OMR treatment (Guimaraes, et al.56, Diaféria,
20 English translation from: Orthod Fr 2019;90:343-370
et al.35,36). The studies by Diaféria, et al.35,36 showed
that at the end of three weeks of therapeutic wit-
hdrawal after intervention the improvement in ESS
remained statistically significant in the OMR and
CPAP groups.
5.1.4. OMR improves adherence to CPAP
treatment
The RCT by Diaféria, et al.36 reported a significant
increase in adherence to CPAP. The mean adherence
was 55% in the control group, 63% in the OMR
group, 30% in the CPAP group and 65% in the com-
bined CPAP + OMR group.
5.1.5. Passive OMR is effective and increases
compliance with reeducation
Passive OMR, with the assistance of a cus-
tom-made orthosis29,63, increases compliance with
reeducation (Huang, et al.63). None of the fifty-four
patients in the active OMR group completed the
one-year therapy follow-up, whereas 88.88% of the
fifty-six patients in the passive OMR group comple-
ted the study63.
Passive OMR resulted in a statistically significant
decrease in snoring intensity, AHI and a significant in-
crease in upper airway (Huang, et al.63, Chuang, et al.28).
5.2. Limitations
A first limitation is the low compliance of adult
and pediatric patients with their active OMR pro-
gram. Another limitation is the difficulty of keeping
participants enrolled in the studies and convincing
them to undergo the complex end-of-study tests
again. This makes it particularly difficult to assess
the persistence of the effects of OMR.
Passive OMR appears to provide an effective res-
ponse to this lack of compliance when based on the
custom-made one-piece mandibular advancement
orthosis designed by Michèle Hervy-Auboiron29,63.
The other two studies (Levrini, et al.77,78), which used a
prefabricated myofunctional appliance for the treat-
ment of paediatric obstructive sleep apnoea, could
not be included in the systematic review because, like
all uncontrolled case series, their level of evidence
was insufficient to lead to recommendations.
Continued attention should be paid to monito-
ring possible adverse effects of devices used for pas-
sive OMR on the dental arches, including vestibulo-
version of the mandibular incisors.
The question arises as to the mode of action of
the orthosis used in the studies by Huang, et al.63
and Chuang, et al.28. Is its real efficiency only linked
to a lingual propulsion and a clearing of the upper
airways by the patient incited to rotate the ball lo-
cated at the anteroinferior end of the orthosis, just
before falling asleep? Is it also and partially related
to the construction of the orthosis with a moderate
mandibular propulsion, at 50% of the maximum
mandibular advancement7,9,141?
Another limitation is the selection of studies. We
included in this review randomised clinical studies
comparing OMR with a placebo or controlled inter-
vention and prospective cohort studies. Due to the
paucity of relevant publications, we also searched
for retrospective cohort studies and included the
2013 study by Guilleminault, et al.53. As with other
retrospective cohort studies (case-control study),
this study has a risk of selection bias4.
The ten studies included in this systematic re-
view had various methodological limitations, main-
ly related to the difficulty of recruiting compliant
patients to participate in OMR treatment and kee-
ping them in the study. For example, in the study by
Chuang, et al.28, the control group had milder forms
of OSA than the treatment group, the sample size
of the control group was significantly smaller than
that of the treatment group, and the adenoids and
tonsils of the control group were larger than those
of the treatment group.
Other limitations relate to the short duration of
OMR programs, for example six weeks in the study
by Neumannova, et al.85, which limits the validity of
the results.
Also, due to the difficulty of recruiting partici-
pants, sample sizes are often small, which leads to
low power of statistical tests.
Finally, OMR is based on an integrative approach
to an exercise programme and therefore does not
allow the specific effects of each exercise on the
overall outcome to be determined.
21Amat P., Tran Lu Y E.
6. Conclusions
Obstructive sleep apnoea syndrome (OSAS) is a
widespread and under-diagnosed condition, making
it a major public health and safety issue.
Orofacial myofunctional reeducation (OMR) has
been shown to be effective in the multidisciplinary
management of OSAS in children, adolescents and
adults and is prescribed at several stages of mana-
gement.
The ten studies included in this systematic review
on the effects of OMR on OSAS show that OMR si-
gnificantly reduces AHI by up to 90.6% in children
and 92.06% in adults. It significantly reduces the
intensity and frequency of snoring, contributes to a
reduction in daytime sleepiness, limits the recurrence
of OSA symptoms after adenotonsillectomy in child-
ren and improves adherence to CPAP therapy.
To address the frequent lack of compliance obser-
ved in OMR programs, some authors have proposed
the use of passive OMR, with the assistance of a cus-
tom-made appliance or prefabricated functional ap-
pliances, the latter with the additional objective of mo-
difying the shape and relationship of the dental arches.
The integration of these devices into OMR proce-
dures requires the practitioner to carefully monitor
the potential adverse effects of the appliances on
the dental arches, particularly the vestibuloversion
of the mandibular incisors.
Passive OMR, with the assistance of a cus-
tom-made ball orthesis, increases compliance with
reeducation, allows a significant decrease in snoring
intensity, AHI and a significant increase in the upper
airway.
Prospective studies with large samples would be
useful to better evaluate the persistence of the ef-
fects of OMR and the possibilities offered by pas-
sive OMR, with the ball mandibular advancement
orthosis designed by Michèle Hervy-Auboiron or
prefabricated functional appliances, to compensate
for the frequent lack of compliance observed during
active OMR treatments.
Links of interest
The authors declare that they have no interest in
the data published in this article.
References
1. Ahuja S, Chen RK, Kam K, Pettibone WD, Osorio RS, Varga
AW. Role of normal sleep and sleep apnea in human memory
processing. Nat Sci Sleep 2018;10:255-269.
2. Aiello KD, Caughey WG, Nelluri B, Sharma A, Mookadam
F, Mookadam M. Eect of exercise training on sleep
apnea: A systematic review and meta-analysis. Respir Med
2016;116:85-92.
3. Amat P. Prise en charge thérapeutique des DAM par rééduca-
tion maxillo-faciale, intégrée à une éducation thérapeutique
du patient: pourquoi, quand, comment ? Rev Orthop Dento
Faciale 2011;45:175-195.
4. Amat P. Dentisterie fondée sur les faits: en omnipratique et en
orthodontie. Paris: Éditions CdP, 2012.
5. Andersen IG, Holm JC, Homøe P. Obstructive sleep apnea in
obese children and adolescents, treatment methods and out-
come of treatment – A systematic review. Int J Pediatr Oto-
rhinolaryngol 2016;87:190-197.
6. Andersen IG, Holm JC, Homøe P. Impact of weight loss
management on children and adolescents with obesity and
obstructive sleep apnea. Int J Pediatr Otorhinolaryngol
2019;123:57-62.
7. Anitua E, Durán-Cantolla J, Almeida GZ, Alkhraisat MH.
Minimizing the mandibular advancement in an oral appliance
for the treatment of obstructive sleep apnea. Sleep Med
2017;34:226-231.
8. Barret RH. One approach to deviate swallowing. Am J Or-
thod 1961;47:726-736.
9. Bartolucci ML, Bortolotti F, Raaelli E, D’Antò V, Miche-
lotti A, Alessandri Bonetti G. e eectiveness of die- rent
mandibular advancement amounts in OSA patients: a sys-
tematic review and meta-regression analysis. Sleep Breath
2016;20(3):911-919.
10. Baz H, Elshafey M, Elmorsy S, Abu-Samra M. e role of
oral myofunctional therapy in managing patients with mild
to moderate obstructive sleep apnea. PAN Arab J Rhinol
2012;2:17-22.
11. Berreto e Silva Pitta D, Farias Pessoa A, Sampaio AL, Nonato
Rodrigues R, Guiot Tavares M, Tavares P. Oral myofunc- tio-
nal therapy applied on two cases of severe obstructive sleep
apnea. Intl Arch Otorhinolaryngol 2007;11:350-354.
12. Biggs SN, Walter LM, Jackman AR, Nisbet LC, Weichard AJ,
Hollis SL, et al. Long-Term Cognitive and Behavioral Out-
comes following Resolution of Sleep Disordered Breathing in
Preschool Children. PLoS One 2015;10(9):e0139142.
13. Blandin PF. Anatomie du système dentaire. Paris, Baillière.
1836.
14. Bonuck K, Parikh S, Bassila M. Growth failure and sleep
disordered breathing: a review of the literature. Int J Pediatr
Otorhinolaryngol 2006;70(5):769-778.
15. Boutron I, Moher D, Tugwell P, et al. A checklist to evaluate
a report of a nonpharmacological trial (CLEAR NPT) was
developed using consensus. J Clin Epidemiol 2005;58:1233-
1240.
16. Butler MP, Emch JT, Rueschman M, Sands SA, Shea SA,
Wellman A, et al. Apnea-Hypopnea Event Duration Predicts
Mortality in Men and Women in the Sleep Heart Health Stu-
dy. Am J Resp Crit Care Med 2019;199(7):903-912.
17. Caloway CL, Diercks GR, Keamy D, de Guzman V, Soose
R, Raol N, et al. Update on hypoglossal nerve stimulation in
children with down syndrome and obstructive sleep apnea.
Laryngoscope 2019 [Epub ahead of print].
18. Camacho M, Certal V, Abdullatif J, et al. Myofunctional the-
rapy to treat obstructive sleep apnea: a systematic review and
meta-analysis. Sleep 2015;38:669-675.
22 English translation from: Orthod Fr 2019;90:343-370
19. Camacho M, Chang ET, Song SA, Abdullatif J, Zaghi S,
Pirelli P, et al. Rapid maxillary expansion for pediatric obs-
tructive sleep apnea: A systematic review and meta-ana- lysis.
Laryngoscope 2017;127(7):1712-1719.
20. Camacho M, Guilleminault C, Wei JM, et al. Oropharyngeal
and tongue exercises (myofunctional therapy) for snoring: a
systematic review and meta-analysis. Eur Arch Otorhinola-
ryngol 2018;275:849-855.
21. Camacho M, Li D, Kawai M, Zaghi S, Teixeira J, Senchak AJ,
Brietzke SE, et al. Tonsillectomy for adult obstructive sleep
apnea: A systematic review and meta-analysis. Laryngoscope
2016;126(9):2176-2186.
22. Carneiro-Barrera A, Díaz-Román A, Guillén-Riquelme A,
Buela-Casal G. Weight loss and lifestyle interventions for
obstructive sleep apnoea in adults: Systematic review and me-
ta-analysis. Obes Rev 2019;20(5):750-762.
23. Carvalho FR, Lentini-Oliveira DA, Prado LB, Prado GF,
Carvalho LB. Oral appliances and functional orthopaedic
appliances for obstructive sleep apnoea in children. Cochrane
Database Syst Rev 2016;10:CD005520.
24. Cassano M, Russo G, Granieri C, Ciavarella D. Modication
of growth, immunologic and feeding parameters in children
with OSAS after adenotonsillectomy. Acta Otorhinolaryngol
Ital 2018;38(2):124-130.
25. Cauhépé J, Fieux J, Coutand A, Bouvet JM. Le rôle mor-
pho- génétique du comportement neuromusculaire. Rev Stom
1955;56(7):535-546.
26. Chauvois A, Fournier M, Girardin F. Rééducation des fonc-
tions dans la thérapeutique orthodontique. Paris: S.I.D., 1991.
27. Cheng SY, Kwong SHW, Pang WM, Wan LY. Eects of an
Oral-Pharyngeal Motor Training Programme on Children
with Obstructive Sleep Apnea Syndrome in Hong Kong:
A Retrospective Pilot Study. Hong Kong J Occup er
2017;30(1):1-5.
28. Chuang LC, Hwang YJ, Lian YC, Hervy-Auboiron M, Pirelli
P, Huang YS, Guilleminault C. Changes in craniofa- cial and
airway morphology as well as quality of life after passive myo-
functional therapy in children with obstructive sleep apnea: a
comparative cohort study. Sleep Breath 2019. doi: 10.1007/
s11325-019-01929-w [Epub ahead of print].
29. Chuang LC, Lian YC, Hervy-Auboiron M, Guilleminault C,
Huang YS. Passive myofunctional therapy applied on children
with obstructive sleep apnea: A 6-month follow-up. J Formos
Med Assoc 2017;116(7):536-541.
30. Costantino A, Rinaldi V, Moa A, Luccarelli V, Bressi F, Cas-
sano M, et al. Hypoglossal nerve stimulation long-term cli-
nical outcomes: a systematic review and meta-analysis. Sleep
Breath 2019. doi: 10.1007/s11325-019-01923-2. [Epub ahead
of print] Review.
31. Dayatt E, Kheirandish-Gozal L, Gozal D. Childhood obs-
truc- tive sleep apnea: one or two distinct disease entities? Clin
Sleep Med 2007;42:374-379.
32. Deeb R, Smeds MR, Bath J, Peterson E, Roberts M, Beckman
N, Lin JC, Yaremchuk K. Snoring and carotid artery disease:
A new risk factor emerges. Laryngoscope 2019;129(1):265-
268.
33. de Felício CM, Silva Dias FV, Folha GA, et al. Orofacial mo-
tor functions in pediatric obstructive sleep apnea and implica-
tions for myofunctional therapy. Int J Pediatr Otorhinolaryn-
gol 2016;90:5-11.
34. de Felício CM, da Silva Dias FV, Trawitzki LVV. Obstructive
sleep apnea: focus on myofunctional therapy. Nat Sci Sleep
2018;10:271-286.
35. Diaféria G, Badke L, Santos-Silva R, Bommarito S, Tuk S,
Bittencourt L. Eect of speech therapy as adjunct treatment to
continuous positive airway pressure on the quality of life of pa-
tients with obstructive sleep apnea. Sleep Med 2013;14:628-635.
36. Diaféria G, Santos-Silva R, Truksinas E, et al. Myofunctio-
nal therapy improves adherence to continuous positive airway
pressure treatment. Sleep Breath 2017;21:387-395.
37. Ehsan Z, Ishman SL. Pediatric Obstructive Sleep Apnea.
Otolaryngol Clin North Am 2016;49(6):1449-1464.
38. Ehsan Z, Ishman SL, Kimball TR, Zhang N, Zou Y, Amin
RS. Longitudinal Cardiovascular Outcomes of Sleep Disorde-
red Breathing in Children: A Meta-Analysis and Systematic
Review. Sleep 2017;40(3).
39. Fédération Française d’Orthodontie. Place de l’orthodontie
dans le dépistage et le traitement du syndrome d’apnées hypo-
pnées obstructives du sommeil (SAHOS) chez l’enfant. Mai
2018.
40. Floras JS. Sleep Apnea and Cardiovascular Disease: An Enig-
matic Risk Factor. Circ Res 2018;122(12):1741-1764.
41. Folha GA, Valera FC, de Felício CM. Validity and reliabi- lity
of a protocol of orofacial myofunctional evaluation for patients
with obstructive sleep apnea. Eur J Oral Sci 2015;123:165-172.
42. Fournier M, Brulin F. Le moment de la rééducation en
O.D.F. Rev Orthop Dento Faciale 1975;9:37-47.
43. Friberg D, Ansved T, Borg K, Carlsson-Nordlander B, Lars-
son H, Svanborg E. Histological indications of a progres- sive
snorers disease in an upper airway muscle. Am J Respir Crit
Care Med 1998;157(2):586-593.
44. Friedman M, Wilson M, Lin HC, Chang HW. Updated sys-
tematic review of tonsillectomy and adenoidectomy for treat-
ment of pediatric obstructive sleep apnea/hypopnea syndrome.
Otolaryngol Head Neck Surg 2009;140(6):800- 808.
45. Frye SS, Fernandez-Mendoza J, Calhoun SL, Gaines J, Sawy-
er MD, He F, Liao D, Vgontzas AN, Bixler EO. Neurocogni-
tive and behavioral functioning in adolescents with sleep-di-
sordered breathing: a population-based, dual-energy X-ray
absorptiometry study. Int J Obes (Lond) 2018;42(1):95-101.
46. Furlan AD, Malmivaara A, Chou R, Maher CG, Deyo RA,
Schoene M, Bronfort G, van Tulder MW; Editorial Board
of the Cochrane Back, Neck Group. 2015 Updated Method
Guideline for Systematic Reviews in the Cochrane Back and
Neck Group. Spine (Phila Pa 1976) 2015;40(21):1660-1673.
47. Garbarino S. Excessive daytime sleepiness in obstructive sleep
apnea: implications for driving licenses. Sleep Breath 2019
[Epub ahead of print].
48. Garliner D. Myofunctional erapy. Saunders, 1971.
49. Ge X, Han F, Huang Y, Zhang Y, Yang T, Bai C, Guo X. Is
obstructive sleep apnea associated with cardiovascular and all-
cause mortality? PLoS One 2013;8(7):e69432.
50. Genta PR, Schorr F, Eckert DJ, Gebrim E, Kayamori F, Mo-
riya HT, et al. Upper airway collapsibility is associated with
obesity and hyoid position. Sleep 2014;37(10):1673- 1678.
51. Gileles-Hillel A, Kheirandish-Gozal L, Gozal D. Biological
plausibility linking sleep apnoea and metabolic dysfunction.
Nat Rev Endocrinol 2016;12(5):290-298.
52. Gugino CF, Dus I. Unlocking orthodontic malocclusions:
an interplay between form and function. Semin Orthod
1998;4(4):246-255.
53. Guilleminault C, Huang YS, Monteyrol PJ, Sato R, Quo S,
Lin CH. Critical role of myofascial reeducation in pediatric
sleep-disordered breathing. Sleep Med 2013;14:518-525.
54. Guilleminault C, Sullivan SS, Huang YS. Sleep-Disordered
Breathing, Orofacial Growth, and Prevention of Obstructive
Sleep Apnea. Sleep Med Clin 2019;14(1):13-20.
55. Guimaraes KC. [Soft tissue changes of the oropharynx in
patients with obstructive sleep apnea]. J Bras Fonoaudiol
1999;1:69-75.
56. Guimaraes KC, Drager LF, Genta PR, Marcondes BF, Lo-
renzi- Filho G. Eects of oropharyngeal exercises on patients
with moderate obstructive sleep apnea syndrome. Am J Respir
Crit Care Med 2009;179:962-966.
23Amat P., Tran Lu Y E.
57. Hakim F, Kheirandish-Gozal L, Gozal D. Obesity and Alte-
red Sleep: A Pathway to Metabolic Derangements in Child-
ren? Semin Pediatr Neurol 2015;22(2):77-85.
58. HAS. Bon usage des technologies de santé. Comment pres-
crire les dispositifs médicaux de traitement du syndrome
d’apnées hypopnées obstructives du sommeil chez l’adulte.
Octobre 2014. https://www.has-ante.fr/upload/docs/appli-
cation/pdf/2014-11/sahos_-_che_de_bon_usage.pdf.
59. Herkenrath SD, Treml M, Priegnitz C, Galetke W, Randerath
WJ. Eects of respiratory muscle training (RMT) in patients
with mild to moderate obstructive sleep apnea (OSA). Sleep
Breath 2018;22(2):323-328.
60. Higgins J, Green S, eds. Cochrane Handbook for Systema-
tic Reviews of Interventions Version 5.0.0 [updated February
2008]. e Cochrane Collaboration; 2008.
61. Hirata RP, Schorr F, Kayamori F, et al. Upper airway collap-
si- bility assessed by negative expiratory pressure while awake
is associated with upper airway anatomy. J Clin Sleep Med
2016;12:1339-1346.
62. Ho AW, Moul DE, Krishna J. Neck Circumference-Height
Ratio as a Predictor of Sleep Related Breathing Disorder in
Children and Adults. J Clin Sleep Med 2016;12(3):311-317.
63. Huang YS, Chuang LC, Hervy-Auboiron M, Paiva T, Lin
CH, Guilleminault C. Neutral supporting mandibular ad-
vance- ment device with tongue bead for passive myofunc-
tional therapy: a long term follow-up study. Sleep Med 2018
S1389-9457(18)30447-7.
64. Huang YS, Guilleminault C. Pediatric Obstructive Sleep
Apnea: Where Do We Stand? Adv Otorhinolaryngol
2017;80:136-144.
65. Ieto V, Kayamori F, Montes MI, et al. Eects of oropha-
ryngeal exercises on snoring: a randomized trial. Chest
2015;148:683-691.
66. Ishii L, Roxbury C, Godoy A, Ishman S, Ishii M. Does Na-
sal Surgery Improve OSA in Patients with Nasal Obstruction
and OSA? A Meta-analysis. Otolaryngol Head Neck Surg
2015;153(3):326-333.
67. Isono S. Obesity and obstructive sleep apnoea: mechanisms
for increased collapsibility of the passive pharyngeal airway.
Respirology 2012;17(1):32-42.
68. John CR, Gandhi S, Sakharia AR, James TT. Maxilloman-
dibular advancement is a successful treatment for obstruc- tive
sleep apnoea: a systematic review and meta-analysis. Int J Oral
Maxillofac Surg 2018;47(12):1561-1571.
69. Johns MW. A new method for measuring daytime sleepiness:
the Epworth sleepiness scale. Sleep 1991;14:540-545.
70. Johnson LR. Control of habits in treatment of malocclusion.
Am J Orthod Oral Surg 1938;24(10):909-924.
71. Kang KT, Koltai PJ, Lee CH, Lin MT, Hsu WC. Lingual
Tonsillectomy for Treatment of Pediatric Obstructive Sleep
Apnea: A Meta-analysis. JAMA Otolaryngol Head Neck
Surg 2017;143(6):561-568.
72. Kayamori F, Bianchini EMG. Eectiveness of orofacial
myofunctional therapy in obstructive sleep apnea in adults:
systematic review. Conference abstract. Sleep Medicine
2017;40,Supplement 1:e34.
73. Kayamori F, Bianchini EMG. Eects of orofacial myofunc-
tional therapy on the symptoms and physiological parame-
ters of sleep breathing disorders in adults: a systematic review.
Rev CEFAC 2017;19(6):868-878.
74. Kuhle S, Urschitz MS. Anti-inammatory medications for
obstructive sleep apnea in children. Cochrane Database Syst
Rev 2011:CD007074.
75. Lee SA, Amis TC, Byth K, Larcos G, Kairaitis K, Robinson
TD, Wheatley JR. Heavy snoring as a cause of carotid artery
athe- rosclerosis. Sleep 2008;31(9):1207-1213.
76. Lee SY, Guilleminault C, Chiu HY, Sullivan SS. Mouth
brea- thing, “nasal disuse” and pediatric sleep-disordered brea-
thing. Sleep Breath 2015;19(4):1257-1264.
77. Levrini L, Salone GS, Ramirez-Yanez GO. Pre-Fabricated
Myofunctional Appliance for the Treatment of Mild to Mo-
derate Pediatric Obstructive Sleep Apnea: A Preliminary Re-
port. J Clin Pediatr Dent 2018;42(3):236-239.
78. Levrini L, Salone GS, Ramirez-Yanez GO. Ecacy of a
Pre-Fabricated Myofunctional Appliance for the Treatment
of Mild to Moderate Pediatric Obstructive Sleep Apnea: A
Preliminary Report. J Clin Pediatr Dent 2018;42(6):475-477.
79. Lian YC, Huang YS, Guilleminault C, Chen KT, Hervy- Au-
boiron M, Chuang LC, Tsai AI. e preliminary results of the
dierences in craniofacial and airway morphology between
preterm and full-term children with obstructive sleep apnea.
J Dent Sci 2017;12(3):253-260.
80. Linz D, Woehrle H, Bitter T, Fox H, Cowie MR, Böhm M,
Oldenburg O. e importance of sleep-disordered breathing
in cardiovascular disease. Clin Res Cardiol 2015;104(9):705-
718.
81. Mason M, Welsh EJ, Smith I. Drug therapy for obstruc-
tive sleep apnoea in adults. Cochrane Database Syst Rev
2013(5):CD003002.
82. Mohamed AS, Sharshar RS, Elkolaly RM, Serageldin M.
Upper airway muscle exercises outcome in patients with obs-
tructive sleep apnea syndrome. Egypt J Chest Dis Tuberc
2017;66:121-125.
83. Moeller JL, Paskay LC, Gelb ML. Myofunctional therapy:
a novel treatment of pediatrics sleep-disordered breathing.
Sleep Med Clin 2014;9:235-243.
84. Moher D, Liberati A, Tetzla J, Altman DG. PRISMA
Group. Preferred reporting items for systematic reviews and
meta-analyses: the PRISMA statement. Journal of Clinical
Epidemiology 2009;62,1006-1012.
85. Neumannova K, Hobzova M, Sova M, Prasko J. Pulmonary
rehabilitation and oropharyngeal exercises as an adjunct the-
rapy in obstructive sleep apnea: a randomized controlled trial.
Sleep Med 2018;52:92-97.
86. Nieto FJ, Peppard PE, Young T, Finn L, Hla KM, Farre R.
Sleep-disordered breathing and cancer mortality: results from
the Wisconsin Sleep Cohort Study. Am J Resp Crit Care Med
2012;186(2):190-194.
87. Noller MW, Guilleminault C, Gouveia CJ, Mack D, Neighbors
CL, Zaghi S, Camacho M. Mandibular advancement for pe-
diatric obstructive sleep apnea: A systematic review and me-
ta-analysis. J Craniomaxillofac Surg 2018;46(8):1296- 1302.
88. Oh S-M, Kim J-H, Kim S-H. Upper airway myofunctional
exercise: A systematic review. JKMA 2019;62:224-230.
89. Omobomi O, Quan SF. Positional therapy in the manage-
ment of positional obstructive sleep apnea-a review of the cur-
rent literature. Sleep Breath 2018;22(2):297-304.
90. Osman AM, Carter SG, Carberry JC, Eckert DJ. Obstructive
sleep apnea: current perspectives. Nat Sci Sleep 2018;10:21-
34.
91. Owens RL, Gold KA, Gozal D, Peppard PE, Jun JC, Dannen-
berg AJ, et al. UCSD Sleep and Cancer Symposium Group.
Sleep and Breathing… and Cancer? Cancer Prev Res (Phila)
2016;9(11):821-827.
92. Parmar A, Baker A, Narang I. Positive airway pressure
in pediatric obstructive sleep apnea. Paediatr Respir Rev
2019;31:43-51.
93. Patinkin ZW, Feinn R, Santos M. Metabolic Consequences
of Obstructive Sleep Apnea in Adolescents with Obesity: A
Systematic Literature Review and Meta-Analysis. Child Obes
2017;13(2):102-110.
24 English translation from: Orthod Fr 2019;90:343-370
94. Perfect MM, Archbold K, Goodwin JL, Levine-Donnerstein
D, Quan SF. Risk of behavioral and adaptive functioning dif-
- culties in youth with previous and current sleep disordered
breathing. Sleep 2013;36(4):517-525B.
95. Philippe J. Histoire de la thérapeutique comportementale. Rev
Orthop Dento Faciale 2012;46:111-117.
96. Pinto JA, Godoy LB, Marquis VW, Sonego TB, Leal Cde F,
Artico MS. Anthropometric data as predictors of obstructive
sleep apnea severity. Braz J Otorhinolaryngol 2011;77:516-
521.
97. Puhan MA, Suarez A, Lo Cascio C, Zahn A, Heitz M, Braen-
dli O. Didgeridoo playing as alternative treatment for obstruc-
tive sleep apnoea syndrome: randomised controlled trial. BMJ
2006;332:266-270.
98. Reckley LK, Fernandez-Salvador C, Camacho M. e eect
of tonsillectomy on obstructive sleep apnea: an overview of
systematic reviews. Nat Sci Sleep 2018;10:105-110.
99. Redline S, Yenokyan G, Gottlieb DJ, Shahar E, O’Connor
GT, Resnick HE, et al. Obstructive sleep apnea-hypopnea and
incident stroke: the sleep heart health study. Am J Resp Crit
Care Med 2010;182(2):269-277.
100. Ricketts RM. Respiratory obstruction syndrome. Am J Or-
thod 1968;54(7):495-507.
101. Rollet D. De l’éducation fonctionnelle à l’occlusion fonction-
nelle. In: Lejoyeux E. et Flageul F. Propositions orthodon-
tiques / Classe II / Situations critiques. Paris: Quintessence,
2011:12-28.
102. Rogers AP. Exercises for the development of muscles of face
with view to increasing their functional activity. Dental Cos-
mos LX 1918;59:857-876.
103. Romette D. Pratique orthodontique quotidienne et compor-
tement neuromusculaire de la région maxillo-faciale. Chir
Dent Fr 1974;44(222):63-66.
104. Rousseau E, Silva C, Gakwaya S, Sériès F. Eects of one-week
tongue task training on sleep apnea severity: A pilot study.
Can Respir J 2015;22(3):176-178.
105. Sanna A. Obstructive sleep apnoea, motor vehicle accidents,
and work performance. Chron Respir Dis 2013;10(1):29-33.
106. Sateia MJ. International classication of sleep disor-
ders-third edition: highlights and modications. Chest
2014;146(5):1387-1394.
107. Sedky K, Bennett DS, Carvalho KS. Attention decit hyperac-
tivity disorder and sleep disordered breathing in pediatric popu-
lations: a meta-analysis. Sleep Med Rev 2014;18(4):349-356.
108. Sharples LD, Clutterbuck-James AL, Glover MJ, Bennett
MS, Chadwick R, Pittman MA, et al. Meta-analysis of ran-
domised controlled trials of oral mandibular advancement de-
vices and continuous positive airway pressure for obstructive
sleep apnoea-hypopnoea. Sleep Med Rev 2016;27:108-124.
109. Sillah A, Watson NF, Schwartz SM, Gozal D, Phipps AI.
Sleep apnea and subsequent cancer incidence. Cancer Causes
Control 2018;29(10):987-994.
110. Simpson L, Hillman DR, Cooper MN, Ward KL, Hunter M,
Cullen S, James A, Palmer LJ, Mukherjee S, Eastwood P. High
prevalence of undiagnosed obstructive sleep apnoea in the ge-
neral population and methods for screening for representative
controls. Sleep Breath 2013;17(3):967-973.
111. Smith DF, Amin RS. OSA and Cardiovascular Risk in Pedia-
trics. Chest 2019;156(2):402-413.
112. Smith DF, Cohen AP, Ishman SL. Surgical management of
OSA in adults. Chest 2015;147(6):1681-1690.
113. Soares EB, Pires JB, Menezes MA, Santana SKS, Fraga J.
Speech therapy and snore and sleep apnea. Rev CEFAC
2010;12:317-325.
114. Steele CM. On the plausibility of upper airway remodeling as
an outcome of orofacial exercise. Am J Respir Crit Care Med
2009;179:858-859.
115. Strang HHW. A Text-book of Orthodontia. Philadelphie:
Lea and Febiger, 1943.
116. Suzuki M, Okamoto T, Akagi Y, Sekiguchi H, Matsui K, Sa-
toya N, et al. 0565 Impact on Oral Myofunctional erapy to
Treat e Patients with Moderate to Severe Obstructive Sleep
Apnea. Sleep 2019;42(Suppl. 1):p. A225.
117. Suzuki H, Watanabe A, Akihiro Y, et al. Pilot study to assess
the potential of oral myofunctional therapy for improving res-
piration during sleep. J Prosthodont Res 2013;57:195- 199.
118. Swinehart EW. Preventive orthodontia. Dental Cosmos
1927:903-910.
119. Talmant J, Deniaud J. Approche actuelle du traitement des
troubles de la ventilation nasale de l’enfant et de l’adoles- cent.
Rev Orthop Dento Faciale 2010;44:285-302.
120. Talmant J, Talmant JC, Deniaud J, Amat P. Du traitement étio-
logique des AOS. Rev Orthop Dento Faciale 2009;43:253-
259.
121. Taranto-Montemurro L, Messineo L, Sands SA, Azarbar-
zin A, Marques M, Edwards BA, et al. e Combination of
Atomoxetine and Oxybutynin Greatly Reduces Obstructive
Sleep Apnea Severity. A Randomized, Placebo-controlled,
Double-Blind Crossover Trial. Am J Respir Crit Care Med
2019;199(10):1267-1276.
122. Taveira KVM, Kuntze MM, Berretta F, de Souza BDM, Go-
dolm LR, Demathe T, et al. Association between obstructive
sleep apnea and alcohol, caeine and tobacco: A meta-analy-
sis. J Oral Rehabil 2018;45(11):890-902.
123. üer U, Ingervall B. Eect of muscle exercise with an oral
screen on lip function. Eur J Orthod 1990;12(2):198-208.
124. Tom C, Roy B, Vig R, Kang DW, Aysola RS, Woo MA, et
al. Correlations between Waist and Neck Circumferences
and Obstructive Sleep Apnea Characteristics. Sleep Vigil
2018;2(2):111-118.
125. Torres-Castro R, Vilaró J, Martí JD, Garmendia O, Gime-
no- Santos E, Romano-Andrioni B, Embid C, Montserrat
JM. Eects of a Combined Community Exercise Program in
Obstructive Sleep Apnea Syndrome: A Randomized Clinical
Trial. J Clin Med 2019;8(3).pii: E361.
126. Trosman I, Trosman SJ. Cognitive and Behavioral Conse-
quences of Sleep Disordered Breathing in Children. Med Sci
(Basel) 2017;5(4). pii: E30.
127. Truesdell B. et F. Deglutition: with special reference to normal
function and diagnosis. Angle Orthod 1937;7(2):90- 99.
128. Tseng WC, Liang YC, Su MH, Chen YL, Yang HJ, Kuo PH.
Sleep apnea may be associated with suicidal ideation in ado-
lescents. Eur Child Adolesc Psychiatry 2019;28(5):635- 643.
129. Tzeng NS, Chung CH, Chang HA, Chang CC, Lu RB, Yeh
HW, et al. Obstructive Sleep Apnea in Children and Adoles-
cents and the Risk of Major Adverse Cardiovascular Events:
A Nationwide Cohort Study in Taiwan. J Clin Sleep Med
2019;15(2):275-283.
130. Uysal T, Yagci A, Kara S, Okkesim S. Inuence of pre-or-
thodontic trainer treatment on the perioral and masticatory
muscles in patients with Class II division 1 malocclusion. Eur
J Orthod 2012;34:96-101.
131. Valbuza JS, de Oliveira MM, Conti CF, Prado LB, de Car-
valho LB, do Prado GF. Methods for increasing upper airway
muscle tonus in treating obstructive sleep apnea: systematic
review. Sleep Breath 2010;14:299-305.
132. Van Tulder M, Furlan A, Bombardier C, et al. Updated me-
thod guidelines for systematic reviews in the Cochrane colla-
boration back review group. Spine 2003;28:1290-1299.
133. Verma RK, Johnson J Jr, Goyal M, Banumathy N, Goswa-
mi U, Panda NK. Oropharyngeal exercises in the treatment
of obstructive sleep apnoea: our experience. Sleep Breath
2016;20:1193-1201.
25Amat P., Tran Lu Y E.
134. Villa MP, Brasili L, Ferretti A, et al. Oropharyngeal exer-
cises to reduce symptoms of OSA after AT. Sleep Breath
2015;19:281-289.
135. Ward CP, York KM, McCoy JG. Risk of obstructive sleep
apnea lower in double reed wind musicians. J Clin Sleep Med
2012;8(3):251-255.
136. Weaver TE, Grunstein RR. Adherence to continuous positive
airway pressure therapy: the challenge to eective treatment.
Proc Am orac Soc 2008;5(2):173-178.
137. Wilson WE. Common perversions of functions of facial mus-
cles with practical methods for their correction. Dental Cos-
mos 1927:351-359.
138. Wishney M, Darendeliler MA, Dalci O. Myofunctional the-
rapy and prefabricated functional appliances: an overview of
the history and evidence. Aust Dent J 2019;64(2):135-144.
139. Wu J1, Zhao G, Li Y, Zang H, Wang T, Wang D, Han D.
Apnea-hypopnea index decreased signicantly after nasal sur-
gery for obstructive sleep apnea: A meta-analysis. Medicine
(Baltimore) 2017;96(5):e6008.
140. Yagci A, Uysal T, Kara S, Okkesim S. e eects of myo-
func- tional appliance treatment on the perioral and mastica-
tory muscles in Class II division 1 patients. World J Orthod
2010;11:117-122.
141. Yanyan M, Min Y, Xuemei G. Mandibular advancement
appliances for the treatment of obstructive sleep apnea in
children: a systematic review and meta-analysis. Sleep Med
2019;60:145-151.
142. Younis A, Baz H, El Maksoud AA. Upper airway exer-
cises in patient with obstructive sleep apnea. Avalable at:
https://pdfs.semanticscholar.org/f5f1/1e5a6fc6c2e30f-
08842158f62eaa5e84abad.pdf
