DOI: 10.

1051/odfen/2009035 J Dentofacial Anom Orthod 2009;12:108-120

Ó RODF / EDP Sciences

Cranio-facial morphology
and obstructive sleep apnea:
the role of dento-facial
orthopedics
Julia COHEN-LÉVY, Philippe CONTENCIN,
Vincent COULOIGNER

ABSTRACT
Orthodontists, thanks to their keen appreciation of facial morphology and their
skills in making functional examinations, are ideally placed to detect breathing
problems in their patients that can require an ENT assessment, with special
attention paid to the evaluation of sleep patterns. After a site of obstruction,
resulting from a soft tissue anomaly and/or a narrowing of the airway passage
through bone, has been established, orthodontists may be called upon to
undertake corrective treatment sometimes in conjunction with a surgical
procedure.

KEYWORDS
Obstructive sleep apnea syndrome
Child
Craniofacial growth
Adeno-tonsillar hypertrophy
Oral appliance.

Address for correspondence:

J. COHEN-LÉVY,
255, rue Saint-Honoré,
75001 Paris.
108
Article available at http://www.jdao-journal.org or http://dx.doi.org/10.1051/odfen/2009035
 CRANIOFACIAL MORPHOLOGY AND OBSTRUCTIVE SLEEP APNEA: THE ROLE OF DENTOFACIAL ORTHOPEDICS

1 - INTRODUCTION
Various estimates ranging from dilating muscles of the pharynx, can
3.2% to 30% have been presented also play a role in causing OSAS or
of the extent of habitual snoring in the intensifying the effects of the mor-
child population and on the 0.7% to phological anomalies in some cases.
10.3% who suffer from obstructive These interrelationships between
sleep apnea syndrome (OSAS), as cranio-facial anatomy and OSAS have
defined by the recurrence of complete frequently been described in adults,
obstruction (apnea) or partial obstruc- but only recently have investigators
tion (hypopnea) of the upper airways taken a lively interest in its occurrence
during sleep1,14,19. in children, with some emphasis being
This pediatric form of OSAS, which placed on the role orthodontists can
can affect newborns as well as ado- play as members of the inter-disciplin-
lescents, has a patho-physiology that ary medical research teams studying
differs from the adult variety, and is this ailment.
particularly hard to detect11,27. While it Collaboration between specialties is
is true that only a polysomnograph not limited to research and diagnosis,
recording can indisputably establish but continues in the application of the
the diagnosis of sleep apnea, the therapeutic sequence with orthodon-
paucity of sleep laboratory services tic appliances like rapid palatal expan-
make this tool available to just a small sion and mandibular advancement
percentage of possible patients. In often being indicated in early treat-
addition, the symptoms of OSAS are ment procedures.
variable, not widely known, and de-
We illustrate this article, which re-
pend on the child’s stage of growth
views contributions published in pe-
and development. (the reader may
diatric OSAS literature on cranio-facial
want to refer to the article on ‘‘How
morphology, with clinical cases trea-
to detect OSAS’’ in this issue).
ted by a team of an ear, nose and
The primary cause of OSAS is an throat (ENT) surgeon, a physician
anatomical narrowing of the upper specializing in sleep disorders, and
airways affecting both the soft tissues an orthodontist. We then discuss the
and cranio-facial structures. Defects in current protocols for the treatment of
local neuro-muscular control, with OSAS, focusing on the participation of
accompanying loss of tone of the specialists in dento-facial orthopedics.

2 - CRANIO-FACIAL ABNORMALITIES AND RISK FACTORS IN OSAS

2 - 1 - Non-syndromic cranio- the mucosa of the upper airways
facial abnormalities (such as mucopolysaccharidosis, the
Prader Willi syndrome, and obesity
In addition to the pathological dis- with fatty infiltration of the pharyngeal
turbances that create a thickening of walls, can reduce the diameter of the

J Dentofacial Anom Orthod 2009;12:108-120. 109

 JULIA COHEN-LÉVY, PHILIPPE CONTENCIN, VINCENT COULOIGNER

ventilatory pathway), examiners may This same group described a facial

also detect certain anatomic obstacles phenotype of children who were at
in an ENT examination. increased risk of obstructive sleep
breathing difficulties, that comprised
34% of the patients with apnea. Some
2 -1 -1 - Nasalfossae and the rhino
of the characteristics of this group were
pharynx a retruded mandible, a hyper-diver-
– choanal stenosis or atresia; gence of the mandible with an increase
– nasal septum anomalies like sco- in the cranio-mandibular, intermaxillary,
liosis, hypertrophy of the inferior turbi- and gonial angles; an elongated face;
nates; and a deep hard palate and a long soft
palate (case 2). These results were
– hypertrophy of adenoidal soft
tissue; confirmed by other authors2,28: while
Rees et al.26 emphasized, instead, the
– pharyngeal tumor. association with micrognathia and ret-
rognathia. Kawashima et al.17 affirmed
that apnea patients with heavily en-
2 - 1 - 2 - Oral cavity, oropharynx larged tonsillar tissues had, in compar-
and hypopharynx ison to control groups, more retruded
– hypertrophy of the palatal and mandibles and a greater increase in
lingual tonsils; lower facial height.
– cranio-facial anomalies, with med- The malocclusions most frequently
iofacial hypoplasia, small or retruded described in association with OSAS are
mandible, which force the tongue to unilateral or bilateral cross bites, open
intrude on the oropharynx13; bites with a low tongue position and a
posterior rotation of the mandible7,32.
– true macroglossia (arterio-veinous
One recent study25, comparing a group
or lymphatic malformation, von Reck-
of children who snored infrequently to a
linghausen neurofibromatosis ...).
group of children suffering from OSAS,
For the majority of children who found significant differences between
suffer from the OSAS syndrome, the the two: children with sleep apnea had
principal etiological factor is hypertro- greater incisal overjet and less overbite,
phy of lymphoid tissue, which can be narrower upper arches, and shorter
treated effectively with tonsillectomy lower arches than children in the con-
or removal of adenoid tissue18. How- trol group.
ever, Guilleminault et al.10, who re-
viewed the records of 400 children
whose tonsils and adenoids had been
2 - 2 - Syndromic abnormalities
removed, have found that in 14.5% of
these cases (case 1) problems with Some of the cranio-facial malforma-
breathing during sleep persisted. In tions that occur in the apnea syndrome
this study the patients who were "non- predispose afflicted children to a col-
responders" to surgery had narrow lapse of the upper airways often mak-
pharyngeal spaces, an under-devel- ing early treatment essential15 . In
oped maxilla, and/or a retruded mand- severe cases, children only a few days
ible. old must be operated on immediately

110 Cohen-Lévy J, Contencin P, Couloigner V. Cranio-facial morphology and obstructive sleep apnea
 CRANIOFACIAL MORPHOLOGY AND OBSTRUCTIVE SLEEP APNEA: THE ROLE OF DENTOFACIAL ORTHOPEDICS

This 8 year-old boy, had persistent OSAS symptoms

despite already having had an adeno-tonsillectomy.
a: on the lateral cephalogram, a nasal septum deviation can be seen together with asymmetry of the base of the nasal
fossae.
b: on the lateral cephalogram, a decrease in the size of the respiratory pathway seems to be localized behind the
tongue, where there is hypertrophy of the lingual tonsillar tissues, or a retro-positioning of the tongue. This patient
seems to have a Class III tendency which would make mandibular advancement inadvisable.
c and d: on intra-oral examination, the palate was seen to be narrow and highly arched, with abnormal transverse
relationships, an anterior infraclusion with insufficient space for the eruption of the upper lateral incisors.
e: the appearance of the boy’s face was consistent with mouth breathing and a rounded lips apart posture at rest. In
order to attempt to reduce the residual IAH, a turbinectomy, ablation of the lower nasal turbinate, and rapid palatal
expansion were planned.

or helped to breathe with mechanical phalo-syndactyl types 1, 5 and 3,

devices. respectively) are characterized by early
calcification of the cranial sutures that
2 - 2 - 1- Syndromes involving the causes hypoplasia of the that cause
maxilla hypoplasia of the mid-face. In these
cases, the facial skeletal appears to
The Crouzon, Apert, Pfeiffer and
demonstrate mandibular prognathism
Saethre-Chotzen syndromes (acro-ce-

J Dentofacial Anom Orthod 2009;12:108-120. 111

 JULIA COHEN-LÉVY, PHILIPPE CONTENCIN, VINCENT COULOIGNER

Cranio-facial morphology in the OSAS "type"

a and b: This patient, an 8 year-old boy, suffered from attention deficit disorder, hyperactivity, and agitated sleep with
constant snoring interrupted by pauses in breathing. In addition to his long face with rings under his eyes, lip closure
was strained, his profile retrusive and reflective of a hyper-divergent typology (Delaire’s architectural analysis
computerized by means of the Tridimä program);
c to e: the intra-oral examination showed a deep, high arch palate, with bilateral cross bite, anterior infraclusion, and a
Class II tendency.

because of the collapse of the naso- the intra-cranial hypertension asso-

maxillary complex and the develop- ciated with sutural calcification, it is
ment of a Class III malocclusion. If the probable that OSAS would be further
defects of these children are treated under-estimated, suspected as it is in
early with neuro-surgery to eliminate 50% of the subjects15,24.

112 Cohen-Lévy J, Contencin P, Couloigner V. Cranio-facial morphology and obstructive sleep apnea
 CRANIOFACIAL MORPHOLOGY AND OBSTRUCTIVE SLEEP APNEA: THE ROLE OF DENTOFACIAL ORTHOPEDICS

a and b: this 4 year-old boy, afflicted with achondroplasia, also had persistent OSAS symptoms despite previous
adeno-tonsillectomy. Visual examination showed facial retrusion, with caved-in nasal appearance, accentuated by
hydrocephaly;
c and d: intra-oral examination revealed a Class II malocclusion with uni-lateral cross bite associated with symmetrical
maxillary insufficiency with lateral deviation.

In trisomy 21 (Downs syndrome) constitutes an additional risk factor for

early calcification of the spheno-occi- OSAS9, affects half of these patients6.
pital suture, hypoplasia of the mid-face Children afflicted with achondropla-
(reduction of palatal length and width), sia, inherited through autosomal
and a diminishing of the cranial base dominance, suffer from dwarfism,
are all found. Relative macroglossia under-development of the maxilla
associated with the syndrome which and the mandible, as well as from

J Dentofacial Anom Orthod 2009;12:108-120. 113

 JULIA COHEN-LÉVY, PHILIPPE CONTENCIN, VINCENT COULOIGNER

functional aberrations that favor ob- obstructs breathing capacity begin-

structive sleep disturbance15,22. In the ning at birth. In the Nager syndrome
case shown in figure 3, the three year- and in the Franceschetti or Treacher-
old patient, came in for consultation Collins syndrome the principal defects
because of persistence of OSAS are maxillo-facial dystosis and under-
problems even though excess tonsillar development of the mandibular rami
tissue had already been removed. On and condyles, the net result being an
examination we noted that his face increase in facial height that promotes
displayed the typical characteristics of oropharyngeal blockage15.
the syndrome, a deep sinking in of the While changes in position can be
bulk of the middle face, associated effective in preventing backward tilt-
with hydrocephaly that had been ing of the tongue, in half the cases the
treated by tapping of the cephalora- obstruction has to be dealt with
chidian fluids (case 3). mechanically or surgically with intuba-
tion, tracheotomy, surgical osteo-dis-
traction of the mandible, or a surgical
2 - 2 - 2- Syndrome involving glossopexy that produces a temporary
the mandible adhesion of the mucosal tissues of
the tongue to the lower lip8. Some
Characteristics of Robin’s syndrome
are symmetrical under-development treatment teams used intra-oral appli-
ances, based on the monobloc that
of the mandible (that diminishes the
height of the rami and the length of Pierre Robin introduced, to unblock
the pharynx mechanically as the
the body and affects the orientation of
the condyles), associated with a pala- mandible is propelled forward4.
tal cleft and glossoptosis that severely

3 – INTERRELATIONSHIP BETWEEN OSAS TREATMENT AND CRANIO-FACIAL

GROWTH
3 - 1 - Continuous Positive compliant patients seldom use the
Airway Pressure (CPAP) device and end up by abandoning it19.
So the continuous airway pressure
Continuous Positive Airway Pres- system is usually reserved for patients
sure is a non-invasive but effective with severe sleep apnea, those who
means of treating OSAS that employs are grossly obese, have anomalous
a nasal mask through which a com- neuro-muscular tone, are waiting for
pressor delivers a positive pressure surgery, or for whom tonsillectomy or
that keeps the airways open no matter adenoidectomy has failed to relieve
what sleeping posture the child the problem.
assumes. The device whose nasal
Most children with sleep apnea do
face mask is supported by head straps
not require long term CPAP treatment
is cumbersome to wear and some
so its effects on maxillary develop-
patients, supported by their parents,
ment are, in theory, limited but they
find it difficult to wear. These non-

114 Cohen-Lévy J, Contencin P, Couloigner V. Cranio-facial morphology and obstructive sleep apnea
 CRANIOFACIAL MORPHOLOGY AND OBSTRUCTIVE SLEEP APNEA: THE ROLE OF DENTOFACIAL ORTHOPEDICS

Familial predisposition to Class III protrusion and development of anterior cross bite after wearing a VPPC mask.
a and b: Karla 5 years old;
c and d: her mother.

are, nevertheless, not always entirely counterbalance the incipient deforma-

inconsequential. Analyses of clinical tion16.
cases have shown that prolonged
application of CPAP forces on the
facial skeletal tends to promote the 3 - 2 - Tonsillectomy,
development of Class III malocclu- adenoidectomy
sions, especially in children who are
predisposed to them (case 4). The A recently conducted systematic
authors believe that a maxillo-facial review with meta-analysis (Bonuck
evaluation of patients at risk is a et al.3, 2009) endeavored to determine
necessity and might show the need the effect of a combination of tonsil-
for the use of a Delaire mask, adjusted lectomy and adenoidectomy on
to the assemblage of the CPAP to growth and its bio-markers. OSAS

J Dentofacial Anom Orthod 2009;12:108-120. 115

 JULIA COHEN-LÉVY, PHILIPPE CONTENCIN, VINCENT COULOIGNER

and other sleep respiratory disorders changed after the nasal obstruction
can produce retardation or a break in was removed thus spontaneously re-
growth rates by interfering with nor- storing contact between upper and
mal nocturnal secretion of the soma- lower lips, allowing tongue position to
totropic hormone, the GH or growth rise, and restoring better head pos-
hormone. ture, all effects that have been repeat-
After surgery, a significant increase edly described since the first studies
in the serum levels of molecular GH on breathing were conducted. But a
mediators has been noted, the insulin- Swiss orthodontist has formulated an
like growth factor I (IGF I) and IGFBP-3 alternative hypothesis in evaluating
(binding protein), along with an in- these results23, asserting that the
crease in height and weight, some- schema of the posterior mandibular
times sufficient, in a "catch-up" effect, rotation in young OSAS patients re-
to restore the patient to normal body sults from a decrease in ramal growth,
growth. development of the condylar cartilage
having slowed down because of a
One team devoted itself to studying
decrease in serum GH, growth hor-
the repercussion of OSAS on cranio-
mone levels.
facial growth31, by comparing over a
five year period the morphology of a
group of children with sleep apnea, 3 - 3 - Rapid palatal expansion
treated with tonsillectomy and adenoi-
dectomy, to a control group of children The work of Linder-Aronson and
with no breathing problems. Before Woodside, cited by Villa30, described
treatment, at an average age of 5.5 the effects of rapid palatal expansion
years, they found significant differ- on breathing even before the identifi-
ences between the apnea patients cation of obstructive sleep apnea in
and the control group, as revealed by children. This orthopedic treatment
cephalometric analyses. The apnea produced a significant increase in the
patients had mandibles that were area of the nasal fossas and a 36.2 to
more inclined posteriorly and maxillae 45% reduction in nasal resistance.
that were more tilted anteriorly. They The creation of additional space for
had an increase in anterior facial the tongue helped, secondarily, to
height, the anterior portions of their open up the oropharynx. In his study-
cranial bases were shorter, their upper Villa30 proposed to evaluate rapid
and lower incisors tilted more lin- maxillary expansion in the treatment
gually, and nasal areas that did not of OSAS, using a sample of children 4
jut as far forward. But five years after to 11 years old, selected in accor-
treatment, the authors found no sig- dance with three criteria:
nificant differences between the two – the existence of a malocclusion,
groups except for length of the cranial with a deep and narrow vaulted palate
base and of the nose both of which associated with overbite in a Class II
remained shorter for the apnea pa- relationship or anterior cross bite;
tients than for the control group. – OSAS symptoms, with habitual
A possible explanation for these snoring and pauses in respiration,
results is that muscular equilibrium sleep that is not restorative, an

116 Cohen-Lévy J, Contencin P, Couloigner V. Cranio-facial morphology and obstructive sleep apnea
 CRANIOFACIAL MORPHOLOGY AND OBSTRUCTIVE SLEEP APNEA: THE ROLE OF DENTOFACIAL ORTHOPEDICS

apnea-hypopnea per hour (AHI) > 1 orthopedics had played alongside ENT
per hour, as measured by poly-somno- surgery Guilleminault et al ) reviewed
graphy; the records of 32 patients, from 4 to 9
– refusal of parents to have tonsil- year old, who needed removal of
lectomy and adenoidectomy per- excess tonsilar and adenoidal tissue
formed on their child. as well as orthodontic treatment and
presented a moderate OSAS condi-
The investigators excluded from
tion, as defined by their having a
their list subjects who were obese,
minimal oxygen saturation level of
had cardio-respiratory or neuro-mus-
90%, and an IAH of less than 20 per
cular problems, had severe cranio-
hour. The children were divided into
facial anomalies, or presented severe
two groups, the first receiving surgical
symptoms. The rapid expander was
treatment before the rapid palatal
soldered to two bands that were
expansion and the other the same
cemented to the maxillary second
therapies in reverse order. The results,
temporary molars and was activated
evaluated by questionnaire and with
by two turns for the first 10 days until
polysomnographic readings taken at
the palatal cusps of the upper molars
the time of diagnosis and between
were in contact with the buccal cusps
three and six months after each
of their mandibular antagonists.
therapeutic procedure had been com-
The extent of expansion achieved pleted, showed that 87.5% of the
was an average of 3.7 + mm in the patients required both types of treat-
canine region and 5.0 + 2.2 mm in the ment. Surgery alone did not correct
molar region. In a 12 month fixed the OSAS completely and in only two
retention period the over-correction patients did the OSAS symptoms
was maintained. The AHI decreased disappear after only orthodontic treat-
significantly after treatment, dropping ment. For two patients the apnea
from 5.8 ± 6.8 to 1.5 ± 1.6 (p = 0.005), condition continued unabated despite
together with an improvement in the their having received both therapies.
index of oxygen saturation and the
In this study, in which some pa-
index of arousal from sleep. It should
tients also received mandibular expan-
be noted that the change in AHI
sion, the maxillary expansion was
differed as a function of the type of
accomplished with the opening of
malocclusion, being greater with chil-
the orthopedic device a quarter of a
dren with Class II overjet and overbite
millimeter per day. Both fixed and
than with children who had cross
removable appliances were used de-
bites. For two patients (14,3%), the
pending on the clinical situation and
rapid palatal expansion had no effect
the preference of the orthodontist.
on AHI, the authors attributing this to
major anatomical blockages, a nasal
septum deviation in one child and 3 - 4 - Mandibular advancement
marked hypertrophy of palatal soft
tissue that was repeatedly re-infected
oral devices30
in the other. Mandibular advancement appli-
In order to evaluate the contribution ances, designed like orthopedic
to the treatment result maxillo-facial activators, free up the pharynx and

J Dentofacial Anom Orthod 2009;12:108-120. 117

 JULIA COHEN-LÉVY, PHILIPPE CONTENCIN, VINCENT COULOIGNER

maintain a propulsive force on the They evaluated the patients with a

mandible, thus keeping palatal and questionnaire, a polysomnograph, and
basal lingual soft tissues in a state of clinical ENT and orthodontic examina-
tension. Just as orthodontists use this tions. No intraoral or cephalometric
type of device to correct Class II X-Rays were taken, in accord with
malocclusions in children and adoles- American recommendations.
cents, therapists can be employ them The oral appliance, made from
to provide symptomatic treatment for alginate impressions, was set to an
snoring and OSAS in adults. advancement reading obtained by
Even if the effectiveness of these having the children bite on wax in a
devices is variable, depending upon forced Class I position, so that the
the degree of mandibular advance- overjet would be corrected and the
ment that can be obtained and the mandible re-centered. In each appli-
severity of any morphological dispa- ance an acrylic pearl was inserted
rities that may be present, oral appli- behind the incisor positions in a way
ances offer selected patients clinical that would encourage proper tongue
relief that is documented by signifi- position. Children were asked to wear
cant changes on polysomnographic the appliance at all times except when
records. eating.
Certain studies have attempted to Of 19 patients treated, 87% had
evaluate their efficacy in treating excessive overbite or a Class II protru-
children with apnea, particularly be- sion. Even though a quarter of the
cause it has been shown that in non- patients abandoned treatment, a six-
obese patients the extent of maxillary month evaluation showed a significant
overjet is associated with the severity decrease in IAH (p = 0.001), while this
of OSAS21. In one randomized clinical breathing variable remained constant
study30, researchers treated 19 chil- in the control group. For 64.2% of the
dren, with an average age of 6.86 treated children the IAH dropped at
years, with an advancement appliance least 50%, showing the effectiveness
for 6 months and compared them to a of the OSAS therapy accomplished in
control group of 13 untreated children parallel to correction of the malocclu-
with an average age of 7.34 years. sion.

4 - CONCLUSION
While orthodontists have made But according to some studies of
evaluation of lip and tongue habits a the morphology of patients with ob-
regular component of the intake ex- structive respiratory difficulties, it
aminations they conduct on potential would appear that when orthodontists
patients, they have not yet routinely assess Class II malocclusions or un-
incorporated an assessment of the derdevelopment or hyper-divergence
quality of sleep and the presence or of upper or lower jaws, it is important
absence of snoring into their pre- that they determine whether or not
treatment record taking. some aspect of obstructive sleep

118 Cohen-Lévy J, Contencin P, Couloigner V. Cranio-facial morphology and obstructive sleep apnea
 CRANIOFACIAL MORPHOLOGY AND OBSTRUCTIVE SLEEP APNEA: THE ROLE OF DENTOFACIAL ORTHOPEDICS

apnea is implicated in the disorder tractive alternatives to or useful ad-

before they formulate a treatment juncts to ENT surgery. More and more
plan. Conclusions of research studies frequently, orthodontists are now
can sometimes be fragile, in view of being asked to begin early treatment
the small samples they consider, but it of patients afflicted with sleep apnea,
is clear that rapid palatal expansion or sometimes for patients still in the
mandibular advancement can be at- temporary dentition stage.

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120 Cohen-Lévy J, Contencin P, Couloigner V. Cranio-facial morphology and obstructive sleep apnea