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British Journal of Oral and Maxillofacial Surgery xxx (2020) xxx–xxx

Review
Oromandibular dystonia: a diagnosis not to miss
D. Britton a,∗ , J.E. Alty b,c , C.J. Mannion b
a University Hospital of Wales, Cardiff and Vale University Health Board
b Leeds Teaching Hospitals NHS Trust
c University of Tasmania, Australia

Accepted 20 February 2020

Abstract

Oromandibular dystonia (OMD) is characterised by sustained or repetitive involuntary movements of the jaw, face, and tongue. People with the
condition may present to their dentist, general practitioner, or a secondary care specialist with non-specific symptoms including jaw or facial
pain, bruxism, subluxations or dislocations of the jaw; fractured teeth or dental restorations, or both; or jaw tremor. Many clinicians are not
aware of the disorder and this can lead to delayed diagnoses, unnecessary complications, and inappropriate treatment. OMD is an important
diagnosis not to miss because referral for specialist management can provide good long-term results. To aid early, accurate diagnosis, this
paper focuses on the key clinical features of the disorder and its dental and medical mimics.
© 2020 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: Dystonia; Oromandibular dystonia; Dental mimics

Introduction tant for maxillofacial surgeons, in particular, to know about

What is dystonia?
Dystonia is defined as a movement disorder that is char-
acterised by involuntary muscle contractions that cause
abnormal movements, postures, or both.1 Oromandibular
dystonia (OMD) is a specific type that presents with sus-
tained or repetitive involuntary movements of the jaw, face,
and tongue. People with OMD may present with non-specific
symptoms (including jaw or facial pain, bruxism, sublux-
ations or dislocations of the jaw; fractured teeth or dental
restorations, or both; or tremulous movements of the jaw) to
their dentist or general practitioner, or may be referred to a
maxillofacial, ENT, or head and neck specialist. It is impor-
∗
Corresponding author.
E-mail addresses: britton danielle@yahoo.co.uk (D. Britton),
jane.alty@utas.edu.au (J.E. Alty), Christopher.mannion@nhs.net
(C.J. Mannion).
the condition because many of the patients referred from the
community to hospital clinics will have had years of mis-
management before finally being given the correct diagnosis.
It is important not to miss dystonia, as it can be managed
successfully with a combination of botulinum toxin injec-
tions, oral medications, physiotherapy, oral appliances, and
occasionally, deep brain stimulation.
Onset is typically between the ages of 45 and 70 years, with
a female:male ratio of around 1.3-2:1.2–5 Prevalence has been
reported as 6.9/100 000, and incidence as 3.3/1 000 000.6
However, the true incidence of dystonia, including OMD, is
generally believed to be under-reported because of missed
and delayed diagnoses.7 This may occur in part because
of clinicians’ limited knowledge, and the wide ranging and
sometimes non-specific clinical presentations. Typically, in
cases of cervical dystonia, the diagnostic delay is between
one and six years.8

https://doi.org/10.1016/j.bjoms.2020.02.018
0266-4356/© 2020 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Britton D, et al. Oromandibular dystonia: a diagnosis not to miss. Br J Oral Maxillofac Surg (2020),
https://doi.org/10.1016/j.bjoms.2020.02.018
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What is the aetiology?
Most cases of adult-onset dystonia are primary and
“idiopathic”1 with no specific cause. Secondary dystonias are
caused by underlying disease and may be inherited (a known
genetic origin) or acquired. Acquired secondary dystonias
include neurodegenerative disorders (such as Parkinson’s dis-
ease and multiple-system atrophy), traumatic and perinatal
brain injury, diseases of the brainstem (for example, hypoxia,
stroke, tumours, or multiple sclerosis), and the side effects of
drugs, particularly antipsychotic medications. It is thought
that most cases of dystonia share a common pathophysiolog-
ical course with three main aspects: loss of inhibition within
the sensorimotor circuit, disrupted sensorimotor integration,
and maladaptive homeostatic plasticity,9 which result in the
muscles becoming overactive.
Classification informs management and prognosis
It is important to classify dystonia by clinical phenotype
(particularly the anatomical distribution and age of onset),
as this guides investigation, management, and prognosis.
The anatomical classification may be described as “focal”
(affecting just one part of the body, such as the lower face),
“segmental” (affecting two contiguous parts such as the lower
face and neck), “multifocal” (affecting two non-contiguous
parts such as the lower face and foot), or “generalised”
(involving the trunk and at least two other sites).
Dystonia should also be classified according to age of
onset, as this gives valuable information about the likely
underlying cause and prognosis. Onset in infancy, childhood,
and adolescence is more likely to be a secondary dystonia,
which includes genetic (particularly the DYT 1 mutation) or
metabolic causes. Cases of early adult onset (21–40 years of
age) tend to be idiopathic; those of late onset in adults (over
40 years of age) are mostly idiopathic, but some may be part
of a neurodegenerative disorder.
When dystonia begins in childhood, it tends to become
generalised – typically starting in a distal limb and then
spreading to affect the whole body. This may or may not
involve a degree of OMD. Adult-onset dystonia is usually
focal, the most common types being cervical dystonia (caus-
ing posturing or tremor of the neck) and upper limb dystonia
(“writers’ cramp” and tremor of the upper limbs). Adult-onset
OMD tends to remain focal or segmental, and generalised
cases are atypical.
Clinical features of OMD
Symptoms
As OMD can masquerade as several other dental and med-
ical diseases it can easily be missed. Commonly reported
symptoms are bruxism and grinding of the teeth, broken and
Please cite this article in press as: Britton D, et al. Oromandibular dystonia: a diagnosis not to miss. Br J Oral Maxillofac Surg (2020),
https://doi.org/10.1016/j.bjoms.2020.02.018
fractured teeth, jaw and facial pain, drooling, subluxation or
dislocation of the temporomandibular joint (TMJ), and invol-
untary movements of the tongue and face.5 Patients may
complain non-specifically of muscular tension and fatigue
within the facial and masticatory muscles,10 and headaches,
depression, anxiety, and sleep disturbance may also be promi-
nent.
Preceding the onset of OMD patients may have a his-
tory of intermittent, involuntary movements involving the
oromandibular musculature that interfere with talking and
eating. There are a number of peculiar features that are typ-
ical of dystonia, and it is useful to ask about them during
the history (in particular “what makes it worse?” and “what
makes it better?”).
Voluntary actions tend to make OMD worse: these may
be local to the oromandibular musculature (for example, a
jaw tremor whilst talking) or in distal parts of the body (for
example, the jaw deviates only when walking). Sometimes
symptoms are task-specific and occur only during certain
activities - for example, only when talking but not when
singing. Paradoxically in some people, voluntary movements
such as chewing or moving the tongue into certain positions
may reduce or temporarily halt their symptoms; this clinical
feature, which is called “geste antagoniste” or sensory trick,
is a characteristic trait of dystonia.1
It is important to ask about occupational history, as pro-
fessional and highly practised woodwind and brass players,
have an increased incidence of OMD, so-called “embouchure
dystonia”. It may present specifically with problems forming
a seal around the mouthpiece of their instrument (“task-
specific”), or may progress so that it manifests during other
activities too – for example, retracting the lips when talking,
or spontaneously grimacing when at rest. 11
Signs
In OMD the contractions of the jaw and facial muscles, the
lingual muscles, or muscles of the tongue, or a combination,
are sustained and patterned (in the same direction). They
are often “twisting” and are usually exacerbated by volun-
tary movement. Sometimes there is an accompanying tremor
of the jaw, tongue, or lips, or hypertrophy of the muscles
affected.1
Abnormal involuntary muscle movements interfere with
normal oral function. They commonly result in “posturing”,
which is seen as involuntary opening or closing of the jaw, or
pulling and twisting the jaw forwards or laterally. The sub-
types depend on the muscles affected and may be classified by
one or more of the following patterns:12 clenching (closing),
opening, deviation, or deflection of the jaw, or involuntary
protrusion of the tongue.
The temporalis, anterior digastric, masseter, and lateral
pterygoid muscles are commonly involved,13 and they can
become tender on palpation. The contractions themselves
may be slow or rapid, episodic (occurring only in certain
positions), or continuous, and can sometimes occur at cer-
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tain times of the day (for example, dopa-responsive dystonia
worsens as the day progresses). This can make diagnosis dif-
ficult. If the patient tries to resist the movements, additional
“overflow” movements may be seen, and to alleviate them
the jaw should be assessed both when relaxed and during
voluntary movement.
Dystonic spasms can present in a variety of ways and may
be seen with nasal contractions, facial grimacing, pursing or
sucking of the lips, dyskinesia of the tongue, retractions of
the corner of the mouth, and platysmal spasms.
OMD is sometimes segmental - associated with dystonia
of the neck or shoulder muscles (cervical dystonia), eyelids
(blepharospasm), upper facial muscles, or larynx (spasmodic
dysphonia). When it occurs with blepharospasm and upper
facial muscle spasm, it is referred to as Meige syndrome.14
Occasionally it may be multifocal – for example, someone
who has had “writers’ cramp” (focal dystonia of the hand)
since young adulthood may present with OMD later in life.
It is useful to ask about previous or longstanding symptoms
of dystonia in other parts of the body, as they may not be vol-
unteered, but their presence increases the likelihood that the
new oromandibular symptoms have been caused by OMD.
Differential diagnosis
There are a number of dental and medical mimics of OMD.
Misdiagnosis may potentially result in incorrect manage-
ment, iatrogenic harm, and the progression of symptoms.
Dental mimics
OMD presents with dysfunction of the oral and facial muscu-
lature as described above. The clinical symptoms and signs
depend on the severity, and the distribution of the affected
muscles.
OMD can be mimicked by features suggestive of a
diagnosis of TMJ disorder, as both can present with pain,
malpositioning of the joint and disc, together with difficulty
with opening or closing the mouth. Reported cases high-
light the presentation of dislocation of the TMJ related to
oromandibular dystonia,15 together with common symptoms
of the jaw being locked open.16 Impaired mastication and a
change in occlusion may also be noted. This can be related
to problems opening and closing the jaw because of dys-
tonic movements, and may present with trismus or lead to
alterations in speech, dysarthria and dysphonia, and difficulty
with swallowing. Difficulties with breathing have also been
reported, but are rare. The unconscious pulling and twisting
of the mandible forwards or laterally because of dystonia may
result in a patient being unable to achieve an intercuspal posi-
tion, and may be incorrectly attributed to an occlusal or TMJ
disorder.17
Dystonic contractions can lead to patients presenting with
symptoms of myofascial pain, headache, muscular tension,
and fatigue. Muscle pain, which results from dystonic activ-
Please cite this article in press as: Britton D, et al. Oromandibular dystonia: a diagnosis not to miss. Br J Oral Maxillofac Surg (2020),
https://doi.org/10.1016/j.bjoms.2020.02.018
3
ity and forceful contractions, may present as facial pain, but
patients may complain of muscular tension or tiredness.18
Cases of presumed bruxism, which leads to attrition of the
teeth and fractured restorations, may in fact be secondary to
dystonia. Successive wearing of a bite guard, the inability of
dentures to “seat” correctly, and damage to the oral cavity
such as the lips, tongue and gingiva, are sometimes also seen
with OMD. The difference between idiopathic bruxism dur-
ing sleep and OMD-induced bruxism is that the latter usually
stops while sleeping. Forceful, involuntary movements of the
jaw can lead to trauma and can damage structures of the oral
cavity, dental restorations, and dentures. They can also result
in excessive dental wear, dental fractures, and trauma to the
lips, gums, and tongue.19 The prevalence of bruxism is higher
in patients with cranial-cervical dystonia.
Symptoms of OMD may be non-specific and may be diag-
nosed as common dental conditions. When assessing patients
it is therefore important to ensure that an accurate history is
taken and that close attention is paid to exacerbating factors
of associated symptoms.
Medical mimics
Neurological movement disorders can sometimes be difficult
to discriminate from OMD, particularly when a jaw tremor
is present. Parkinson’s disease, for example, can present with
a tremor that affects the jaw, chin, mouth, or tongue, and
can lead to the teeth clicking together or difficulty with
eating. Observing when the tremor occurs may help to dis-
criminate between them. Parkinson’s manifests with a “rest”
tremor (when the mouth is closed, or relaxed open) and often
stops with actions such as talking or wide opening of the
mouth, but OMD usually worsens with voluntary movements.
Accompanying clinical features such as slowed walking, an
asymmetrical tremor of the limbs at rest, and reduced facial
expression, all point towards Parkinson’s too. Occasionally,
essential tremor includes a tremor of the jaw or lip, which
tends to worsen as the mouth is opened voluntarily (a pos-
tural or action tremor) and is relieved when the mouth is
closed.20 By definition, essential tremor will be accompanied
by a bilateral upper limb tremor.
It is well known that OMD may worsen with stress and
anxiety, but this association sometimes results in it being
wrongly diagnosed as a functional neurological disorder or
somatisation. Furthermore, the clinical phenotype of OMD
often includes depression and anxiety, and the prominence
of these symptoms in some cases can lead to the misdiagno-
sis of a purely psychiatric disorder or stress-related illness.
Repetitive brief and non-rhythmic movements of the orofa-
cial musculature can be seen in people with tics. These can
be associated with vocalisations and are more common in
childhood or when associated with Tourette syndrome.
Drug-induced movement disorders can manifest with dys-
tonia, including OMD, or tardive dyskinesias. Both tend to be
caused by drugs that block dopamine (such as antipsychotic
and antiemetic drugs), particularly in elderly patients.21
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Table 1
Key learning points.
Oromandibular dystonia (OMD) is characterised by sustained or repetitive involuntary movements of the jaw, face, and tongue, resulting in abnormal
postures and sometimes a tremor.
A thorough history that focuses on exacerbating and relieving factors, and specific actions, is important to discriminate OMD from dental and medical
mimics.
Examination should be done with the jaw in a relaxed state and also during voluntary movement, as OMD signs may not be present all the time.
Temporalis, anterior digastric, masseter, and lateral pterygoid muscles are commonly involved and may be hypertrophied and tender on palpation.
OMD can be managed successfully with a combination of botulinum toxin injections, oral medications, physiotherapy, oral appliances, and
occasionally, deep brain stimulation.

Drug-induced OMD can occur shortly after treatment has
begun or may appear after many years of treatment. It may
present with a patterned mixture of sustained (causing pos-
turing similar to trismus) and intermittent (causing tremulous
movements of the tongue and jaw) muscle contractions. It can
usually be reversed when the drug is stopped. In contrast,
tardive dyskinesia may persist, or may even present, years
after the drugs have been stopped, and is difficult to treat. It
is therefore important to ask about a history of taking these
drugs. The movements of tardive dyskinesia are characterised
by repetitive oral, facial, and lingual movements that resem-
ble grimacing, chewing, smacking of the lips, and complex
protrusive or writhing movements of the tongue.22
Recreational drug use, particularly of amphetamine,23 has
been associated with serious oral and dental side effects
including uncontrollable clenching of the jaw and pain in the
TMJ that mimic OMD. This drug acts as a stimulant of the
central nervous system and an activator of the sympathetic
nervous system, and may lead to extensive grinding of the
teeth, and clenching of the jaw or muscle trismus, or both.
that worsen with voluntary actions. Presentation to a variety
of healthcare professionals, such as dentists, general prac-
titioners, neurologists, or maxillofacial or ENT specialists,
means that the disorder can easily be misdiagnosed as another
dental or medical mimic. To aid the control of symptoms and
to limit complications, patients with OMD should be referred
to specialist oral and maxillofacial surgeons and neurologists
for further management. Key learning points are shown in
Table 1.
Conflict of interest
We have no conflicts of interest.
Ethics statement/confirmation of patients’ permission
Not required.

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https://doi.org/10.1016/j.bjoms.2020.02.018
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