J Indian Sot Pedo Prev Dent March 2002

16

Anaesthetic considerations of temporomandibular joint ankylosis

with obstructive sleep apnoea : a case report
Shah F. Ra, Sharma R. K.b, Hilloowalla R. N.C, Karandikar A. D.d Mumbai.

T.N.MEDlCAL

COLLEGE AND B. Y.L. NAIR CHARITABLE HOSPITAL

The peroperative management of a 4 l/2 yr old child

having bilateral recurrent temporomandibular joint (TMJ)

ankylosis and obstructive sleep apnoea (OSA) is presented

here. From the anaesthesiologists point of view, the patient
demands special considerations as regards difficulty in
intubation, high sensitivity to all central depressant drugs,
awake extubation, and post-operative maintenance of a
clear airway./t is to be concluded that these conditions call
for strict adherence to the basic principles of airway
management and close observation extending into the
post-operative
period.
1

(J Indian Sot Pedo Prev Dent 2002; 20:1:16-20)

KEY WORDS : Temporomandibular joint ankylosis,
Obstructive sleep apnoea,Blind awake intubation,Postoperative airway maintainence.
.

T he

Temporo-mandibular joints (TMJ) are highly

specialized bilateral joints comprising an articulation
between the cranium and the mandible. Its ankylosis
presents an unusual problem. Amongst its various
causes, the commonest are inflammation and trauma.
In children, it usually starts insidiously after trauma,
which is most often missed and noticed only when the
mouth opening is severely restricted as a result of
which the child cannot bite and masticate solid food.
TMJ ankylosis, especially with mandibular hypoplasia
presents a serious problem for airway management.
The relatively difficult problem becomes even more grave
in the pediatric age group because of their small mouth
opening, near total trismus and uncooperativeness while
securing the airway in the awake state. In children
with longstanding bilateral TMJ ankylosis during the

a.

Lecturer

b. Associate Professor
c. Senior Registrar
d.

Senior

Registrar,

Department

of Anaesthesiology.
-.
_*
:

in
#

active growth phase, a hypoplastic and retrognathic

mandible with severe bird face deformity is noted and
hence (OSA) is obstructive sleep apnoea is relatively
more common and a potentially fatal associated disorder.
The children pose the following anaesthetic risks2.
1.

Extreme sensitivity to all central depressant drugs;

avoid sedation and opioids.

2. Peri-operative risk of apnoea, desaturation and

dysarrythmias; careful and intensive monitoring
pre-operatively extending to the postoperative
period.
3. Difficulty in securing the airway; as mentioned
earlier awake intubation is a safer approach.
4. Post extubation desaturation and hypoventilation;
extubate only when the patient is fully awake and
out of the effect of all the anaesthetic agents.
Monitor in intensive care unit or high dependency
unit.
5. Associated cardiovascular and respiratory
complications of OSA like right ventricular failure
and car pulmonale, systemic and pulmonary
hypertension, polycythaemia and its adverse
influences on anaesthetic management: careful
planning of anaesthetic management and readiness
to treat complications.
CASE REPORT :
A 4 l/2 yr old male child weighing 14 Kg, was
admitted with a history of inability to open his mouth
since early infancy with no obvious history of birth
trauma or infection. Limited mouth opening was noticed
at the age of 7-8 months, which was investigated and
subsequently operated on at the age of 2 years for
release of bilateral TMJ ankylosis. At the time of surgery,
elective tracheostomy was done, and the patient was
successfully weaned off tracheostomy within two days
of surgery. Post-operatively, mouth opening was possible
with effort (not voluntary), but it gradually decreased to
almost nil in the next few months after surgery.

J Indian Sot Pedo Prev Dent March 2002

18

the treatment was abandoned.

of laryngeal anatomy.

Ten days later all carious teeth were restored using

GIC and stainless steel crowns in the Pedodontia
department. The child was discharged from the hospital
after two weeks. Regular follow-ups were maintained.
Repeat sleep studies were done two months later and
showed marked improvement in the sleep indices (AHI1) and LSAT-80%).

Tracheostomy is obviously associated with severe

morbidity and mortality and longterm side effects and
hence was reserved as the last option in case of
emergency. In this case, a tracheostomy could have
been techincally difficult to perform because of the
previous tracheostomy scar and fibrosis. A manual jet
ventilation kit with variable outlet pressure regulator
valve at our disposal, in order to tide over an emergency
crisis, if mask ventilation failed and tracheal intubation
was not possible.Fibreoptic laryngoscopy guided
intubation is an ideal alternative but is quite expensive
and a variety of sizes are needed in pediatric patients.
So it was decided to use blind awake nasal intubation,
which has disadvantages like possibility of trauma and
bleeding from the upper airway.

DISCUSSION :
The causes of TMJ ankylosis may be congenital
(forceps delivery), traumatic, infectious, or of unknown
etiology. Other rarer causes also exist. The younger
patients have greater tendency towards re-ankylosis.
The difficult intubation in TMJ ankylosis in children
results from severe trismus and associated mandibular
hypoplasia. When TMJ ankylosis occurs during the
growth of the mandible, varying degrees of facial
deformities result. Since these children grow with facial
asymmetry, the position of the larynx may be altered.
Classically, bird-faced deformities with convex facial
profiles4 (Fig.2) have been described in chronic long
standing TMJ ankylosis characterized by micrognathic
mandible with receding chin and steep occlusal plane
(Fig. 3). Maxilla, soft tissues enveloping the mandible
and suprahyoid muscles are all secondarily affected.
Oropharyngeal airway is narrowed secondary to
shortening of the mandibular rami with narrowing of the
space between the mandible angles. All these features
command
careful assessment
of the airway
preoperatively. There are no predictors of difficult
intubation such as Malampatti sign (with was not possible
to elicit in this case), Patils sign or Wilsons criteria,
etc3. in pediatric patients. Our main guide was clinical
history, x-ray neck (AP and lateral view) and sleep
studies.
Blind nasal intubation, retrograde intubation, fibreoptic guided intubation or tracheostomy are the various
alternatives for securing the airway. All these have their
own advantages and disadvantages and hence, any
particular technique has to be selected keeping in mind
the patients age, clinical condition, availability of
equipment and morbidity during the procedure.
In this case, although a retrograde intubation kit
was kept ready, it was not tried as the first option, in
view of minimal mouth opening (hence difficulty in
retrieving the catheter per orally), age of the patient
and past history of tracheostomy with likely distortion

The bird face deformity is responsible for OSA in

these patients. The cause of airway obstruction is
multifaceted and includes structural encroachment on
the oropharyngeal and hypopharyngeal lumen, sub
atmospheric intrapharyngeal
pressure a n d t h e
h y p o t o n i c i t y o f t h e o r o p h a r y n g e a l muscles5, thus
apposing the tongue and the posterior pharynx6 (Fig.4).
Anaesthetic agents, sedatives and opioid analgesics
worsen OSA by several mechanisms. First and most
important, they tend to reduce pharyngeal muscle tone
and thus increase the likelihood of upper airway collapse.
Secondly, they reduce the arousal and ventilatory
response to hypoxaemia and hypercapnia. Therefore,
use of any sedatives during the pre-induction period.
was avoided. Once the airway was secured, I.V.
midazolam (0.03) mg/kg) and pentazocine were given
in minimal doses. As the surgical procedure was
reasonably long, the patient was completely out of the
effect of these drugs by the end of the surgery.
lntraoperative course was uneventful. The risk of airway
obstruction is maximum risk in the postoperative period.
The risk of airway obstruction is maximal because of
superimposition of the residual effects of the anaesthetic
drugs and the airway edema on a preexisting precarious
airway. Therefore, immediate extubation of these patients
may prove to be riskya. In this case, the child was
wide awake and not tolerating the endotracheal tube,
was completely out of the effects of the neuromuscular
relaxants, as confirmed by PNS, and was breathing
adequately. Hence the patient was extubated on the
operating table itself. However after 15.20 minutes the

17

Shah F.R., Sharma R.K., Hilloowalla R.N., Karandikar A.D

On admission to our institute he was evaluated and

investigated completely and was found to have the
following findings :
1. History of snoring during sleep.
2. Restricted opening with inter-incisor gap of about
5mm (Fig. 1).
3. Retuded and deficient mandible and chin with
bird face(Fig. 2,3).
4. Scar of previous tracheostomy.

5. Scar of previous surgery in the preauricular area.

6. Bilateral accentuated antegonial notching (Fig.
3).
7. Normal neck movements.
8. No palpable lymph nodes.
9. ENT examination showed no abnormalities, with
right nostril more patent than the left.
10. CT Scan showed bilateral TMJ ankylosis.
11. X-Ray neck (AP and lateral views) showed no
evidence of compression or deviation of the trachea.
12. Pre-operative sleep studies were done (mini
polysomnography) a n d i t s h o w e d a A p n o e a
Hypopnoea Index (AHI) of 13 with lowest 0,
saturation (LSAT) of 65% indicating that the child
had moderate OSA2.
13.

Lateral cephalogram was not done pre-operatively,

because for a 4 l/2 year child in the mixed dentition
phase, standardization of measurements is not
possible.

Remaining biochemical investigations were within

normal limits. Pre-operatively the child was referred to
the Pedodontia department for opinion. Anterior root
piece extractions were done. The patient was visited
often in the ward to establish a good rapport and to
explain the anaesthesia technique.
On the day of surgery, after taking an informed consent
from the patients parents, the patient was premeditated
with intravenous glycopyrrolate, ranitidine, metoclopramide
and hydrocortisone, 30 minutes prior to the procedure.
No sedatives were administered pre-operatively.
Xylometazoline decongestant drops were placed in both
the nostrils. Monitors like pulse-oximeter, cardioscope,
capnometer and peripheral nerve stimulator (PNS) were
attached. The patient was catheterized after induction
to monitor the urine output. The child was preoxygenated
adequately and blind awake nasotracheal intubation
,,, . .
1.
I
. -_

using a 4.5 no. plain red rubber endotracheal tube was

done after giving bilateral superior laryngeal nerve blocks
(1.5~~ of 1.5% adrenalised xylocaine on each side)
and injecting ICC of 2% plain xylocaine intratracheally.
Endotracheal intubation was confirmed using
capnometry, bilateral chest auscultation and reservoir
bag movement. Subsequently I.V. propofol and I.V.
pancuronium bromide were administered. Anaesthesia
was maintained using 0, + N,O + traces of halothane
and long acting nondepolarising muscle relaxant with
PNS as a guide I. V. pentazocine ( after intubation
was achieved) and a rectal diclofenac suppository were
administred for analgesia. I. V. midazolam (0.03) mg/
kg) was also given as a sedative and amnesic agent.
Bilateral TMJ ankylosis release with interpositional
gap arthroplasty using temporalis myofacial graft and
bilateral extraoral coronoidectomy was done. The blood
loss during surgery was within tolerable limits and the
patient remained hemodynamically stable throughout
the procedure.
At the end of the procedure, all anaesthetic agents
were switched off. Recovery from muscle relaxation
was confirmed by PNS and the patient was reversed
using I.V. atropine and neostigmine. Before extubating.
laryngoscopy was done and an attempt was made to
visualize the larynx. Vocal cards could not be visulized
since the patient had Cormack-Lehane grade-IV
laryngoscopic view3. The endotracheal tube was
subsequently removed after thorough suctioning.
The patient was wide-awake following extubation.
After 15-20 minutes, end tidal carbon dioxide (ETCO,)
started rising. The patient was put in lateral position.
A tongue stitch was attempted, but failed as patient
was already wide-awake and there was a risk of bleeding.
Hence, a nasopharyngeal airway was placed in the
right nostril and humidified 0, was given using T-piece
connected to Jackson-Rees circuit. The patient was
shifted to a high dependency unit with pulse-oximetric
and capnographic monitoring. Patient maintained 0,
saturation with adequate CO, washout while breathing
spontaneously throughout the night. Mouth opening
without any assistance was 3.5 cm. Patient was started
on jaw stretching exercises from the third postoperative
day.
Nasal continuous positive airway pressure (nCPAP)
was tried at night to prevent tongue fall and OSA, but
the child did not tolerate the nCPAP mask and hence

Shah F.R., Sharma R.K.. Hilloowalla R.N., Karandikar AD.

Fig. 1 : Preoperative

frontal

view

showing

interincisor

gap

of

Fig. 2 : Preoperative

profile

view

showing

bird

face

deformity.

5mm.

Fig. 3 : Postoperative

lateral

cephalogram

showing

retrognathia.

Fig. 48: The likely position when asleep in obstructive apnoea.

The tongue and palate are completely occluding the upper
airway and the very negative pressure in the hypopharynx
in compounding the collapse.

Fig. 4A: The awake airway

Fig. 4C: The beneficial effects of nCPAP in acting as a pneumatic

splint to the airway.

20

J Indian Sot Pedo Prev Dent March 2002

ETC02 started to rise in the supine position. The patient

was turned to a lateral position and a nasopharyngeal
airway was inserted which ensured continuous oxygen
supplementation and prevented tongue fall. Other options
available were to keep the endotracheal tube in situ for
24 hours postoperatively and allow the child to self
extubate once completely out of the effects of the
anaesthetic agents or use airway exchange catheters
to facilitate reintubation if requiredg.
The various treatment options available for OSA are
either medical (nCPAP), or surgical (maxillary and
mandibular advancement along with hyoid suspension
& genioplasty)lO. Nasal CPAP acts as a pneumatic
splint (Fig.4) and thus prevents upper airway collapse2.
It is also known to reduce mucosal edema. This patient
did not tolerate nCPAP mask and hence the procedure
had to be abandoned.
Simultaneous surgical correction of the ankylosis
and the facial deformities have the advantage of a single
operation and the psychological benefit to the patient
in view of immediate improvement in the facial appearance
and mouth opening. However, in our case, due to age
consideration this was not attempted because:
1. As the child grows, the rapid mandibular growth
may compensate and correct the retrognathra on
its own to some extent.
2.

Simultaneous mandibular osteotomy with distraction

appliances would make the bone unstable and
hence prevent postoperative vigorous jaw
physiotherapy, which is a must to prevent reankylosis especially in children.

Therefore corrective surgery was planned for a later

date.
From the above discussion, we can conclude that
patients with TMJ ankylosis and OSA pose a difficult
anaesthesia management problem that requires careful
planning with attention to every minute detail.

REFERENCES
1.

Vas L.,

Sawant P. A review of anaesthetic technique in

15 paediatric patients with temporomandibular joint ankylosis.

Paediatric Anaesthesia 2000; 10 : l-8.
2.

Nader N. Boushra. Anaesthetic management of patients

with sleep apnoea. Can. J. Anaesthesia 1996; 43 : 599616.

3.

Benumof J. L. Management of the difficult adult airway.

Anaesthesiology 1991; 75 : 1087-1110.

4.

El-Sheikh M. M., Medra A. M., Warda M. H. Bird face

deformity

secondary

to

bilateral

Temporomandibular

joint

ankylosis J. Craniomaxillofacial surgery 1996; 24 : 96103.

5.

Remmers J. E., Anch A. M. Sleeping and breathing. What

to do for lack of both Editorial commentary. Arch Intern
Med 1981; 141 : 989.

6.

Orr W. C., Martin R. J. Obstructive Sleep Apnoea

associated

with tonsillar hypertrophy in adults. Arch Intern Med 1981;

141 : 990-992.
7.

Hanning C. D. Obstructive Sleep Apnoea. Br. J Anaesth

1989; 63 : 477-488.

8.

Tierny M. N., Pollard B. J., Doran 8. R. Obstuctive Sleep

Apnoea. Anaesthesia 1989; 44 : 235-237.

9.

Miller K. A., Harkin C. P.,

Bailey P. L., Post-operative

tracheal extubation. Anaesth Analg 1995; 80 : 149-172.

10.

Cohen S. R.,

Simms C., Burstein F. D., Thomsen J.,

Alternatives to tracheostomy

in infants and children with

Obstuctive Sleep Apnoea. J of Paediatric Surgery 1999;

34 : 182-187.
11.

Dodds C. Sleep Apnoea and Anaesthesia. Recent advances

in Anaesthesia and Analgesia 1994; 18 : 179-195.

Reprint requests to :
Dr. Falguni Shah,
133, Princess Street,
5/5th Floor,
Mumbai-400

002.

(Maharashtra,

INDIA)