Neurogenic temporomandibular joint dislocation treated ... - SOMICS
Neurogenic temporomandibular joint dislocation treated ... - SOMICS
Neurogenic temporomandibular joint dislocation treated ... - SOMICS
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<strong>Neurogenic</strong> <strong>temporomandibular</strong> <strong>joint</strong> <strong>dislocation</strong> <strong>treated</strong> with<br />
botulinum toxin: report of 4 cases<br />
Olga Vázquez Bouso, MD, a Gabriel Forteza González, MD, PhD, b Jens Mommsen, MD, DDS, a<br />
Víctor Gumbao Grau, MD, a Javier Rodríguez Fernández, MD, a and Mario Mateos Micas, MD, a<br />
Tarragona, Spain<br />
DEPARTMENT OF ORAL AND MAXILLOFACIAL SURGERY, UNIVERSITY HOSPITAL JOAN XXIII<br />
Many patients suffer recurrent episodes of <strong>temporomandibular</strong> <strong>joint</strong> (TMJ) <strong>dislocation</strong> due to an excess of<br />
muscle contraction or spasticity in the depressor muscles of the jaw. The manual repositioning using the Nelaton<br />
maneuver is the first treatment. Occasionally, it may be necessary to use sedation or general anesthesia to achieve the<br />
desired muscle relaxation. In case of recurrence, surgical treatment is indicated. One nonsurgical method of treatment<br />
is the local infiltration of botulinum toxin type A. We present 4 cases of recurrent TMJ <strong>dislocation</strong> in patients suffering<br />
from conditions of neurologic origin, with considerable motor deterioration, <strong>treated</strong> with local infiltration of botulinum<br />
toxin type A. In conclusion, the injection of botulinum toxin type A is an effective method in cases of neurogenic TMJ<br />
<strong>dislocation</strong>, with low morbididty and side effects, improving patients’ quality of life. (Oral Surg Oral Med Oral Pathol<br />
Oral Radiol Endod 2010;109:e33-e37)<br />
The anterior <strong>dislocation</strong> of the <strong>temporomandibular</strong> <strong>joint</strong><br />
(TMJ) is by far the most frequent form. It occurs when<br />
one or both mandibular condyles are displaced in front of<br />
the temporal eminence. It may be reducible when it returns<br />
spontaneously to the glenoid cavity, or nonreducible<br />
when 1 or 2 condyles remain dislocated. In this position,<br />
the mouth remains open due to the action of the elevator<br />
muscles with or without lateral deviation, depending on<br />
whether the <strong>dislocation</strong> is unilateral or bilateral.<br />
Dislocation of the TMJ is generally of unknown origin,<br />
with several theories put forward to explain its onset.<br />
Some disorders of collagen metabolism, such as ligamentous<br />
hyperlaxity and Ehler-Danlos syndrome might be<br />
related. However, there is a group of patients who suffer<br />
recurrent episodes of TMJ <strong>dislocation</strong> due to an excess of<br />
muscle contraction-spasticity in the muscles depressing<br />
the lower jaw. These patients are frequently associated<br />
with severe disorders of the central nervous system, with<br />
motor and cognitive deterioration, thus contraindicating<br />
surgery under general anesthesia. Daelen defined these<br />
cases of TMJ <strong>dislocation</strong> as neurogenic, 1 to distinguish<br />
them from traumatic injuries. Some degenerative neurologic<br />
disorders are accompanied by repeated bouts of TMJ<br />
<strong>dislocation</strong>, such as Parkinson disease or multiple sclero-<br />
a<br />
Attending, Maxillofacial Surgical Department, Joan XXIII Hospital,<br />
Tarragona, Spain.<br />
b<br />
Chairman, Maxillofacial Surgery Department, Joan XXIII Hospital,<br />
Tarragona, Spain.<br />
Received for publication Oct 1, 2009; accepted for publication Oct<br />
21, 2009.<br />
1079-2104/$ - see front matter<br />
© 2010 Published by Mosby, Inc.<br />
doi:10.1016/j.tripleo.2009.10.046<br />
sis, as well as posttraumatic cerebral palsy, particularly<br />
those occurring with spasticity and orofacial or oromandibular<br />
dystonia.<br />
The treatment of acute episodes, in the case of nonreducible<br />
TMJ <strong>dislocation</strong>, consists of the manual repositioning<br />
using the Nélaton maneuver. 2 Occasionally,<br />
in cases of inveterate TMJ <strong>dislocation</strong>, it may be necessary<br />
to use sedation and even general anesthesia to<br />
achieve the desired muscle relaxation. 3<br />
In cases of recurrence, surgical treatment is indicated<br />
using various techniques. These are basically divided<br />
into 2 groups:<br />
1) Eminectomy (Myrhaug technique). 4<br />
2) Interposition techniques (redirecting the tendon of<br />
the temporal muscle, alloplastic or bone graft, <strong>dislocation</strong><br />
fracture of the zygomatic arch). 5-8<br />
One nonsurgical method of treatment that has been<br />
described is the injection of sclerosing substances in the<br />
area adjacent to the <strong>joint</strong>, including arthroscopic techniques.<br />
9<br />
In those patients suffering neurogenic TMJ <strong>dislocation</strong>,<br />
the reiteration of the <strong>dislocation</strong> produces local<br />
pain and severe eating difficulties. This, together with<br />
the inconvenience of repeated visits to emergency centers,<br />
causes anxiety in patients and their relatives. In<br />
these cases, TMJ <strong>dislocation</strong> occurs through a loss of<br />
coordination of the muscles in the jaw and hypertonia<br />
of the muscles depressing the lower jaw.<br />
.The use of local treatment with botulinum toxin type A<br />
might be effective in such patients. The toxin produces<br />
temporary denervation on the muscles depressing the<br />
lower jaw, thus preventing excessive displacement of the<br />
e33
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e34 Vázquez Bouso et al. March 2010<br />
condyle when opening. Botulinum toxin type A is indicated<br />
for the treatment of a focal dystonias, 10,11 and it is<br />
effective to treat episodes with excess of muscular activity.<br />
We decided to use botulinum toxin type A to treat 4<br />
cases of recurrent TMJ <strong>dislocation</strong> of neurologic origin<br />
after approval by the ethics committee and authorization<br />
of each case by the Ministry of Health.<br />
CASE REPORTS<br />
We present 4 cases of recurrent TMJ <strong>dislocation</strong> in patients<br />
suffering from conditions of neurologic origin <strong>treated</strong> with<br />
local infiltration of botulinum toxin type A.<br />
Case 1<br />
A 26-year-old male who suffered severe head and brain<br />
trauma in 2003 as the result of a traffic accident. After the<br />
accident, he presented multiple foci of brain contusions, with<br />
subarachnoid bleeding in the third and lateral ventricles, the<br />
right thalamus, and the right occipital cortex. These injuries<br />
led to the presence of chronic subdural haematomas. He also<br />
suffered diaphyseal fracture of the right humerus, right shutter<br />
ring, right acetabulum, and sacrum.<br />
The clinical consequence of these injuries was the onset of<br />
right-side hemiparesia, predominantly brachial, as well as<br />
movement dystonia, particularly marked in the orofacial<br />
sphere, and widespread spasticity. The most striking manifestation<br />
was the presence of recurrent episodes of <strong>temporomandibular</strong><br />
<strong>dislocation</strong> when eating, yawning, or speaking,<br />
with a strong tendency to relapse and requiring multiple<br />
treatments (simple manual reduction under sedation, temporary<br />
intermaxillary fixation under general anesthesia) over the<br />
course of 2 years.<br />
For this reason, after obtaining the family’s written consent,<br />
he was injected in September 2007 with 25 UI botulinum<br />
toxin type A (Botox; Allergan, Irvine, Calif), under<br />
electrical control (Botox Injection Amplifier) in each of the<br />
external pterygoid muscles. The technique used in this patient<br />
was that described by Daelen et al. 12 With the patient in a<br />
sitting position, the location of the sigmoid notch and the<br />
condyle of the jaw were determined on either side by palpation.<br />
Next, the needle 13 connected to the electrical amplifier<br />
was inserted 1 cm in front of the condyle, obliquely backwards,<br />
down, and in. When the patient was asked to open his<br />
mouth, slight movements were made with the needle to capture<br />
the signal corresponding to the contraction of the external<br />
pterygoid muscle. Then, after aspiration, the contents of the<br />
syringe were injected. No side effects were noted during<br />
administration or in the subsequent interval. Approximately<br />
10 days after the treatment, the episodes of TMJ <strong>dislocation</strong><br />
ceased. This allowed the patient to significantly improve in<br />
independence to carry out physiologic functions and in his<br />
ability to communicate (Fig. 1).<br />
In October 2008, he was <strong>treated</strong> again, after reappearance<br />
of the TMJ <strong>dislocation</strong>. Forty UI of botulinum toxin type A<br />
(Botox) was injected into both external pterygoid muscles and<br />
10 UI into the anterior faces of both digastric muscles. After<br />
1 week had elapsed, moderate dysphagia appeared, accompanied<br />
by oropharyngeal candidiasis, probably secondary. He<br />
Fig. 1. Electrical amplifier and needles for injection.<br />
was <strong>treated</strong> with a soft diet and Nystatin mouthwash, and the<br />
condition disappeared in 2 weeks. Once more, the episodes of<br />
TMJ <strong>dislocation</strong> disappeared, and he remained in remission<br />
for 10 months to the time of writing.<br />
All of these treatments were administered on an outpatient<br />
basis.<br />
Case 2<br />
A 72-year-old female patient, diagnosed as having Alzheimer<br />
and Parkinson diseases, had severe intellectual impairment<br />
and widespread spasticity. Since May 2008, she had<br />
presented repeated bouts of inveterate TMJ <strong>dislocation</strong> associated<br />
with eating, yawning, and even sleeping, leading her to<br />
be repeatedly taken to our hospital’s Emergency Unit for its<br />
reduction, on occasions under general anesthesia, in view of<br />
the associated spasticity that hindered simple manual reduction,<br />
even under sedation.<br />
In January 2009, after obtaining the family’s consent, the<br />
patient was subjected to general anesthesia in the operating<br />
theater by means of nasotracheal intubation.<br />
No systemic muscle relaxants were used. With the assistance<br />
of the electrical amplifier, per the technique described<br />
by Moore and Wood, 13 the mandibular opening reflex was<br />
explored by striking the chin with a reflex hammer (Fig. 2).<br />
The capture of the amplified electrical signal produced by this<br />
reflex allowed the localization of the infiltration point. Twentyfive<br />
units of Botox was injected into both external pterygoid<br />
muscles (Fig. 3). No immediate or late-onset side effects were<br />
observed. The patient was discharged the next day.<br />
Over the next 7 months, the patient did not present any<br />
episode of TMJ <strong>dislocation</strong>. The patient’s quality of life and<br />
autonomy notably improved during this period, according to<br />
the testimony of her relatives.<br />
Case 3<br />
A 88-year-old woman presented to us for recurrent bilateral<br />
TMJ <strong>dislocation</strong> that had started several months before<br />
the initial referral. She had diagnosed Alzheimer and Parkinson<br />
diseases with widespread spasticity.<br />
In February 2009, after obtaining the family’s written<br />
consent, she was subjected to sedation in the operating the-
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Volume 109, Number 3 Vázquez Bouso et al. e35<br />
Fig. 2. Localization of the infiltration point with electrical<br />
amplifier as per the technique described by Moore.<br />
Fig. 3. Botulinum toxin type A injection by electrical amplifier.<br />
ater. We injected 25 UI Botox into both external pterygoid<br />
muscles.<br />
Over the next 6 months, the patient did not present any<br />
episode of TMJ <strong>dislocation</strong>.<br />
Case 4<br />
A 23-year-old male suffering from Steinert disease (myotonic<br />
dystrophy) began to develop recurring episodes of TMJ<br />
<strong>dislocation</strong> in October 2007. The <strong>dislocation</strong> occurred while<br />
performing minimal movements of mouth opening, including,<br />
e.g., daily dental hygiene. He frequently had to use our<br />
emergency department for reduction, making his normal life<br />
impossible.<br />
In March 2009, after giving written consent, he was <strong>treated</strong><br />
by injection of 25 UI Botox into each lateral pterygoid muscle<br />
under local anesthesia. The muscle was localized using the<br />
Plexygon 7501.31 nerve stimulator (Vygon) connected to a<br />
injection needle (Locoplex 17°, 23 gauge, 35 mm; Vygon).<br />
Fig. 4. Nerve stimulator and needle for injection.<br />
Applying a stimulation of 1 mA, frequency 2 Hz, 300 �s, it<br />
was assumed that the tip of the needle penetrated the muscle<br />
when a rhythmic movement of the mandible occurred in<br />
synchronicity with the stimulation (Fig. 4).<br />
No side effects were noted. The patient presented with a<br />
TMJ <strong>dislocation</strong> episode 5 months after treatment, and a new<br />
dose of Botox was administered.<br />
DISCUSSION<br />
Botulinum toxin type A is a double-chain protein,<br />
150 kD in weight, produced by Clostridium botulinum,<br />
an anaerobic bacterium responsible for botulism. It<br />
prevents the release of acetylcholine into the synaptic<br />
cleft, temporarily blocking neuromuscular transmission.<br />
14 It acts selectively on the peripheral cholinergic<br />
nerve endings 15 to produce muscle relaxation, diminished<br />
compression of the muscle vessels, and occasionally<br />
a reduction in the concentration of excitatory neuropeptides.<br />
16<br />
Botulinum toxin type A was first indicated for the<br />
treatment of strabismus in patients �12 years old,<br />
blepharospasm, hemifacial spasm, and associated focal<br />
dystonias. Later, new indications were approved, such<br />
as cervical dystonia (for the reduction of signs and<br />
symptoms of spasmodic torticollis), dynamic equine<br />
foot deformity caused by spasticity in children �2<br />
years old with cerebral palsy, arm spasticity in patients<br />
who have suffered a cerebrovascular accident, 11 and,<br />
recently, axillary hyperhidrosis.<br />
In the literature, there is an increasing number of<br />
reports on the applications of botulinum toxin for the<br />
treatment of disorders of the TMJ. Bakke et al. 17 described<br />
its use in severe clicking of the TMJ associated<br />
with anterior disc displacement, with good results.<br />
The treatment with botulinum toxin type A is contraindicated<br />
in the case of hypersensitivity to any of the
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e36 Vázquez Bouso et al. March 2010<br />
components in its formulation and when there are widespread<br />
disorders of muscle activity; e.g., it should not<br />
be used in patients with myasthenia gravis or Eaton<br />
Lambert syndrome, when aminoglycoside antibiotics or<br />
spectinomycin are or will be administered, or when<br />
there is inflammation or infection at the injection site. 18<br />
In 1997, Moore and Wood 13 and subsequently<br />
Daelen 12 described the treatment of TMJ <strong>dislocation</strong><br />
with botulinum toxin in patients affected by recurrent<br />
TMJ <strong>dislocation</strong> and neurologic disorders. The technique<br />
used by the authors is based on the work of Brin<br />
et al., 19 who first described the injection of botulinum<br />
toxin type A into the external pterygoid muscles under<br />
electromyographic control for the treatment of oromandibular<br />
dystonia.<br />
In the present cases, it is reasonable to assume that<br />
during injection there is a certain diffusion of botulinum<br />
toxin into the pterygoid space, affecting not only<br />
the lateral but also the medial pterygoid muscle. The<br />
denervation of the medial pterygoid muscle is compensated<br />
by other masticatory muscles, such as the masseter<br />
or the temporal muscles. The denervation of the<br />
lateral pterygoid muscle is not compensated, thus preventing<br />
movements of the condyle that lead to <strong>dislocation</strong>.<br />
In 1998, Daelen described 5 cases of neurogenic<br />
TMJ <strong>dislocation</strong> <strong>treated</strong> with botulinum toxin, with<br />
scant undesirable effects. 20 Only 1 of our cases presented<br />
some side effects: the appearance of transitory<br />
dysphagia, probably due to the toxin spreading from the<br />
anterior fasces of the digastric muscles. From the perspective<br />
of our current experience, we do not consider<br />
it to be necessary to administer the drug in this location,<br />
because there is no need for it and because of the<br />
possible onset of dysphagia. Other injection techniques<br />
include intraoral route with electromyografic guide, as<br />
described by Martínez-Pérez and García Ruiz-Espiga. 21<br />
Case 2 presented particularly difficult characteristics,<br />
in view of the impossibility of injecting the toxin into<br />
the patient while conscious, owing to a lack of cooperation<br />
resulting from the underlying disorder. For this reason, we<br />
used the technique described by Moore and Wood in<br />
1997, 13 which allows the medication to be administered in<br />
a controlled fashion under general anesthesia. However,<br />
the outcome in this case was the most satisfactory, as<br />
indicated by the improvement in the patient’s quality of<br />
life. The greatest difficulty lies in not using systemic<br />
muscle relaxant drugs and in observing the mouth opening<br />
reflex, which can easily be confused with the mouth<br />
closure reflex (masseter reflex). Probably, at present, the<br />
most effective technique is the use of the nerve stimulator,<br />
as is described in case 4.<br />
The doses used (between 25 and 40 UI) depend on<br />
the type of drug (Botox in this series), because the other<br />
preparation available on the market, Dysport, is �4<br />
times less active than Botox, 22 although there is no<br />
fixed dose relationship. In view of the scarcity of literature<br />
on the subject, there are no references about the<br />
dose per individual or the titration of botulinum toxin<br />
type A. Therefore, the doses used in this series can be<br />
considered to be valid, although, as mentioned above,<br />
they depend on the preparation used and different formulations<br />
are not interchangeable.<br />
The irrelevance of side effects, together with the<br />
dramatic results on these patients’ quality of life, makes<br />
TMJ <strong>dislocation</strong> treatment with botulinum toxin type A<br />
an extraordinarily useful resource as an alternative to<br />
well tried surgical techniques that are difficult to apply<br />
in patients with sever neurologic disorders. Moreover,<br />
the etiology of TMJ <strong>dislocation</strong> in these cases is confirmed<br />
by the efficacy of botulinum toxin type A. There<br />
is no limit on the number of doses if a minimum<br />
interval of 3 months is respected between applications.<br />
Respecting this time interval minimizes the formation<br />
of neutralizing anti–botulinum toxin type A antibodies,<br />
thus maintaining its efficacy without limit of time.<br />
In conclusion, the injection of botulinum toxin type<br />
A in the external pterygoid muscles under instrument<br />
control is an effective method in cases of neurogenic<br />
TMJ <strong>dislocation</strong>, with scant morbidity and side effects,<br />
improving patients’ quality of life and their interaction<br />
with their surroundings.<br />
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22. Sampaio C, Ferreira JJ, Simoes F, Rosas MJ, Magalhaes M,<br />
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Reprint requests:<br />
Olga Vázquez Bouso, MD<br />
Department of Oral and Maxillofacial Surgery<br />
University Hospital Joan XXIII<br />
C/ Dr.Mallafré Guasch n°4<br />
43007 Tarragona<br />
Spain<br />
vbolga@hotmail.com