International Journal of Pediatric Otorhinolaryngology (2008) 72, 385—389

www.elsevier.com/locate/ijporl

Primary mandibular distraction for management

of nocturnal desaturations secondary to
temporomandibular joint (TMJ) ankylosis
P. Anantanarayanan *, Vivek Narayanan, R. Manikandhan,
Dharmendra Kumar

Department of Oral & Maxillofacial Surgery, Meenakshiammal Dental College & Hospital,
Alappakkam Main Road, Maduravoyal, Chennai 600095, India

Received 27 September 2007; received in revised form 26 November 2007; accepted 26 November 2007

KEYWORDS Summary
Distraction;
Objectives: Mandibular advancement is a proven method in the management of
Mandibular distraction;
obstructive sleep apnoea syndrome (OSAS) which may manifest as sleep disturbances
Sleep apnoea;
with nocturnal desaturations during sleep (NDS). The purpose of this study is to
TMJ ankylosis
evaluate the role of primary osteo-distraction prior to ankylosis release in pediatric
patients, diagnosed with NDS secondary to temporomandibular joint (TMJ) ankylosis.
Methods: Three patients in the age group of 8—12 years diagnosed with OSAS
secondary to TMJ ankylosis underwent primary osteo-distraction for mandibular
advancement. They were evaluated pre- and post-operatively using radiographs,
over night pulse oximetry, and subjective evaluation of their sleep patterns.
Results: All the three patients showed significant improvement in their saturation
levels with a mean oxygen saturation of 94.66%. There was marked reduction in their
snoring and sleep/awakening patterns. The mean advancement of the mandible in the
three patients was 13.8 mm.
Conclusion: Primary mandibular distraction is an effective method of correction of
nocturnal desaturations during sleep in patients with TMJ ankylosis.
# 2007 Elsevier Ireland Ltd. All rights reserved.

1. Introduction systemic disease. This condition involving the fusion

TMJ ankylosis is a disorder of the joint which occurs
secondary to trauma, local or systemic infection, or
* Corresponding author. Tel.: +91 44 26212366.
E-mail address: anantparameswaran@yahoo.com
(P. Anantanarayanan).
of the mandible and the temporal bone gives rise to
a myriad of deformities of the facial skeleton. The
features presented include hypomobility of the
mandible, retrogenia, micrognathia, dental maloc-
clusion, change in the cant of the maxillary occlusal
plane, airway embarrassment and recurrent upper
airway tract infections [1,2]. The presence of

0165-5876/$ — see front matter # 2007 Elsevier Ireland Ltd. All rights reserved.
doi:10.1016/j.ijporl.2007.11.015
386 P. Anantanarayanan et al.

retruded mandible and micrognathia in these

patients create a narrowing of the oro-pharyngeal
space with mechanical obstruction to respiration,
more so in supine position and during sleep. This
process forms complex syndrome of apnoeic epi-
sodes with significant reduction in the mean oxygen
saturation levels during sleep and secondary cardiac
and respiratory problems [1,3,4].
Mandibular distraction in the last decade has
been accepted as a good modality in managing
the hypoplastic mandible, without the necessity
for bone grafts or complex osteotomies [5—7].
One of the main advantages of DO involves distrac-
tion of soft tissue along with lengthening of the
bone. Because of the gradual and controlled move-
ment of the bone, the surrounding soft tissues are
‘‘recruited’’ or ‘‘stretched’’ simultaneously [7].
Numerous studies now demonstrate the active use
of distraction in the management of compromised
airway and sleep apnoea in the paediatric popula-
tion [4,6,7,8].
Mandibular distraction has been used successfully
to manage the facial deformities associated with
TMJ ankylosis [1,8—11]. However the simultaneous
use of mandibular distraction with ankylosis release
has significant disadvantages: (1) improper outcome
of distraction due to unpredictable vector manage-
ment and (2) physical interference of the distraction Fig. 1 Pre-operative frontal photograph demonstrating
process, to active post-operative physiotherapy gross facial asymmetry.
after ankylosis release [1,8,11]. The use of genial
distraction as a modality to overcome these diffi- 2.2. Methods
culties may not be useful in the case of pediatric
patients where there is influence of mixed dentition 2.2.1. Evaluation
and the probability of further mandibular growth The patients were evaluated pre- and post-opera-
[1]. Hence we decided to manage the primary pro- tively with parental questionnaires and pulse oxi-
blem of sleep apnoea first, with a stage 1 mandib- metry. The questionnaire included queries related
ular advancement by distraction which would to presence of snoring, awakening episodes during
increase the patency of the oro-pharyngeal airway, sleep and day time somnolence. The parents were
followed by a second stage release of the TMJ also questioned regarding the activity of the chil-
ankylosis. dren during the day. The SpO2 of the patients were
recorded and the oxygen desaturation index (ODI)
was calculated. The ODI of >4% (ODI4) [12] was
2. Subjects and methods taken into consideration as recommended for
assessment of severity of sleep apnoea in pediatric
2.1. Subjects population. A comparison of pre- and post-operative
evaluations was done and recorded. Multiple
Three patients with TMJ ankylosis in the age group recordings were done for the patients and the mean
of 8—12 years who presented with complaints of was calculated for reliability.
snoring during sleep, episodes of night time awa-
kening and day time somnolence were included in 2.2.2. Mandibular distraction and ankylosis
this study during the period from July 2006 to 2007. release
Two of them had bilateral TMJ ankylosis and one had All the patients underwent a thorough radiographic
unilateral involvement (Fig. 1). All of them showed evaluation using lateral and frontal cephalograms.
gross facial deformity with severe mandibular retro- They were evaluated for oro-pharyngeal airway
gnathism. All three were assessed with question- patency using McNammara’s airway analysis and
naires to the parents, and over night pulse oximetry. by Grummon’s analysis for planning the quantum
Primary mandibular distraction for management of NDS 387

Fig. 2 Frontal photograph showing distractor in situ.

and vector of distraction. A monofocal external pin-
fixated distraction device was used for the proce-
dure. The distraction was performed with a latency
period of 4 days and activation for the required
quantum of movement and consolidation for 8
weeks (Fig. 2). The post-operative assessment was
carried out at the end of the consolidation period.
All patients underwent the second stage of surgical
release of ankylosis release followed by physiother-
apy and rehabilitation 1 month after consolidation
(Figs. 3 and 4).
3. Results
The assessment revealed a significant reduction in
the ODI4 of all the patients and an overall improve-
Fig. 3 Post-operative frontal photograph after stage 2
demonstrating good facial symmetry.
ment in the mean oxygen saturation as indicated in
Table 1.
There was a 4% improvement in the mean oxygen
saturation levels and a noteworthy reduction of the
mean ODI4 by 24.2.
The mean advancement of the mandible
achieved by distraction was 13.8 mm.
All patients showed cessation of snoring and
decrease in their night time awakening episodes
and day time somnolence.
All the post-operative lateral cephalograms
showed a significant increase in the oro-pharyngeal
airway as shown (Figs. 5 and 6).

Table 1 Table demonstrating subjects and results

Patient Unilateral/ Pre-operative Post-operative Pre-operative Post-operative Mandibular
bilateral mean O2 mean O2 mean ODI4 mean ODI4 advancement
ankylosis saturation saturation
Left side Right side
1 Unilateral 91 95 24.8 0 16 —
2 Bilateral 90 94 31.6 8.1 14 15
3 Bilateral 91 95 22.5 4.2 12 12
Mean 90.66 94.66 26.33 4.1 13.8
388 P. Anantanarayanan et al.

Fig. 5 Pre-operative lateral cephalogram demonstrat-

ing constricted pharyngeal airway.

Fig. 4 Frontal photograph demonstrating optimal mouth

opening.

4. Discussion

Micrognathia and retrognathia are common features

seen in a number of congenital and acquired cra-
niofacial anomalies including Treacher Collins syn-
drome, Hemifacial Microsomia, Nager syndrome,
Pierre Robbin sequence and TMJ ankylosis [13].
Numerous surgical options have been described
for the management of the hypoplastic mandible
of which distraction osteogenesis has gained signifi-
cant acceptance in terms of the predictability of the
outcome and lower morbidity post-surgically [8,11].
Mandibular distraction was proposed by McCarthy in
1992 and has been popularised in the management
of the hypoplastic mandible by Carls et al. [14] and
Morovic and Monasterio [6]. Carls et al. have dis-
cussed the successful use of mandibular distraction
in children with a 7-year follow-up. Monasterio
reports two groups of patients in the pediatric
age group describing the use of distraction in the
management of obstructive sleep apnoea in man-
dibular hypoplasia and shows favourable results [3].
Cohen reports a remarkable reduction in the
respiratory distress index in his series of patients Fig. 6 Post-operative lateral cephalogram demonstrat-
with the use of distraction osteogenesis [5]. ing remarkable improvement in the pharyngeal airway.
Primary mandibular distraction for management of NDS
Unlike the other congenital craniofacial anoma-
lies enumerated above, TMJ ankylosis is an
acquired condition which produces a severe degree
of retrognathia and micrognathia which may lead to
OSAS [1,13]. The management of the deformed
mandible in ankylosis has been done using different
procedures. The gold standard in children was the
costo-chondral graft which was known for its high
post-surgical morbidity and unpredictable growth.
Distraction osteogenesis has become a recent addi-
tion in the surgical management of the mandibular
micrognathia and is fast gaining popularity [8].
Though there are good results reported in litera-
ture with the use of distraction for TMJ ankylosis, it
also has major drawbacks. There have been two
protocols followed in TMJ ankylosis: (1) simulta-
neous distraction with ankylosis release and (2)
release of ankylosis followed by distraction in the
second stage [8—11]. The major disadvantages of
these techniques are (1) undesirable outcome of
distraction, due to active mobilisation of the cal-
lous during physiotherapy after ankylosis release
and (2) the unpredictable movement of the small
proximal stump of the mandible, after the osteot-
omy for the distraction. Generally this proximal
stump shows a lateral and posterior movement
and fails to dock to the recontoured glenoid fossa
[8,11].
The primary problem of sleep disturbance and
NDS in our group of patients shifted the focus to
the correction of the mandibular hypoplasia
before the release of the joint. In this group of
patients mandibular distraction was performed
prior to release of the joint and showed excellent
results in the management of the sleep disorder.
The patients were reviewed and recalled after
3 months and checked for consolidation of
the distracted bone. The patients then under-
went release of the ankylosis followed by rehabi-
litation.
This technique of primary mandibular distraction
followed by ankylosis release has shown good and
predictable results in the management of the noc-
turnal desaturation during sleep, without compro-
mising the out come of the distraction process or the
ankylosis release.
Available online at www.sciencedirect.com
389
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