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Case Report

Oromandibular Dystonia: A Rare Clinical Entity

Shailaja B, Suprakash Chaudhury, Shalesh Rohatgi1, Daniel Saldanha

Departments of Psychiatry Oromandibular dystonia (OMD) is a chronic, disabling focal dystonia involving

Abstract
and 1Neurology,
Dr. DY Patil Medical College,
Dr. DY Patil Vidyapeeth,
Pune, Maharashtra, India
masticatory, facial, pharyngeal, lingual, and lip muscles. OMD interferes with
normal orofacial functions, such as deglutition and speech leading to impaired
quality of life. Cosmetic disfiguration and social embarrassment faced by these
patients owing to the perturbing nature of movements leads to anxiety, depression
and social isolation. There are very few reported cases of OMD in Indian
literature as the disorders are often misdiagnosed. We present two cases of OMD
who were initially misdiagnosed but later responded well to appropriate therapy.
A comprehensive evaluation and a holistic approach are crucial for the prompt
management of OMD.

Received: 03‑08‑2018
Keywords: Blepharospasm‑plus syndrome, botulinum toxin type A, geste
Accepted: 16-01-2019 antagoniste, oromandibular dystonia

Introduction and ill‑fitting dentures. Task‑specific OMDs were seen in

O romandibular dystonia (OMD) is a disorder of
movement affecting the lower part of the face
and the jaw. It is characterized by the involuntary,
paroxysmal, sustained, or intermittent movements of jaw
and tongue with facial grimacing produced by spasms
of the masticatory, facial, pharyngeal, lingual, and lip
muscles of variable severity. These movements can be
patterned, twisting, or tremulous. OMD is classified as
jaw opening, jaw closing, jaw deviating, lingual, and
mixed types. OMD presenting with blepharospasm once
known as Brueghel syndrome or Meige’s syndrome is
currently included in blepharospasm‑plus syndrome due
to the imprecise nature of the eponyms. OMD generally
appears at middle age or beyond. It is twice more
common in women than in men. More than one‑third
of the patients with OMD have dysphagia, dysarthria,
impaired salivation and few suffer from dental attrition,
headache, and facial pains. Cosmetic disfiguration and
social embarrassment faced by the patients owing to
the perturbing nature of movements leads to anxiety,
depression and social isolation thus hampering the quality
of life. OMD is either primary reflecting a dysfunction in
the basal ganglia‑thalamo‑cortical circuits or secondary
to medications, essential tremors, Parkinson’s disease,
Huntington disease, neuroacanthosis, Wilson disease,
Lesch‑Nyhan syndrome, oromandibular‑facial trauma,
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wind instrument players, auctioneers, and bingo callers.
The most common secondary type is tardive dystonia,
developing as a side effect of long‑term treatment with
antipsychotic drugs. Due to high‑clinical variability and
rare occurrence, OMD poses diagnostic and therapeutic
challenges.[1‑8] In this case series, we present two cases
of OMD.
Case Reports
Case A
A 54‑year‑old female presented with gaping of mouth,
spontaneous, repetitive, spasmodic movement of the
lower jaw, facial grimaces, bouts of belching, frequent
blinking and difficulty in opening eyes fully for the
last 2 years. Due to the inability to close the mouth
completely, she used to wrap a scarf tightly around her
face to close the mouth. She used to have spontaneous,
repetitive, painless movements of the lower jaw muscles
throughout the day, and bouts of belching slowly
subsiding over 5–6 h which were socially embarrassing.
Though she could not exercise control over the jaw
Address for correspondence: Dr. Suprakash Chaudhury,
Department of Psychiatry, Dr. DY Patil Medical College,
Dr. DY Patil Vidyapeeth, Pimpri,
Pune ‑ 411 018, Maharashtra, India.
E‑mail: suprakashch@gmail.com
This is an open access journal, and articles are distributed under the terms of the Creative
Commons Attribution‑NonCommercial‑ShareAlike 4.0 License, which allows others to
remix, tweak, and build upon the work non‑commercially, as long as appropriate credit is
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For reprints contact: reprints@medknow.com

DOI: How to cite this article: Shailaja B, Chaudhury S, Rohatgi S, Saldanha D.

10.4103/mjdrdypu.mjdrdypu_123_18 Oromandibular dystonia: A rare clinical entity. Med J DY Patil Vidyapeeth
2019;12:370-2.

370 © 2019 Medical Journal of Dr. D.Y. Patil University | Published by Wolters Kluwer - Medknow
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Shailaja, et al.: Oromandibular dystonia
movements or belching, it used to subside to a certain
extent, whenever she engaged herself in conversations.
She used to find difficulty in swallowing and chewing
due to which, liquids were preferred over solids. She
would use the straw to drink. Over the span of 2 years,
she had lost almost 8 kg of her body weight due to
altered food habits. Her speech had several pauses and
breaks. Although she could fetch a fairly good amount
of sleep as movements used to temporarily disappear on
falling asleep, sleep onset would often be delayed due
to the troublesome nature of the jaw movements. She
would blink often. At times, it would be difficult for her
to open her eyes fully, so she would manually open her
eyes. The aforementioned symptoms had an insidious
onset and progressive course. Over the last 2 years, she
had consulted multiple physicians and psychiatrists and
was diagnosed with the dissociative motor disorder and
was tried on a various combination of selective serotonin
reuptake inhibitors, benzodiazepines and low‑dose
antipsychotic medication with no improvement.
Case B
A 76‑year‑old female presented with complaints of
spontaneous, repetitive, painless spasmodic movements
of lower jaw throughout the day, pursing of lips and
bouts of belching lasting for about half an hour for
the last 1½ year. Her speech had long pauses. The jaw
movements used to subside to a certain extent whenever
she would engage in conversations and would disappear
completely with sleep. Despite multiple consultations
with physicians over a year, there was no improvement
leading to psychiatric referral.
In both the subjects, there was no significant stressor
in temporal relation to the onset of illness. Bouts of
belching experienced by the subjects were thought
to be secondary to aerophagia caused by open mouth
breathing. They were distressed by the burdensome
nature of the symptoms, altered food habits, difficult
articulation, cosmetic disfiguration, and social
embarrassment secondary to it and were disappointed
with the undue delay in the diagnosis and treatment.
There were no primary or secondary gains. There
was no appreciable drug history before the onset.
No relevant past or family history of any medical/
psychiatric illness could be noted. Premorbid personality
was well adjusted. Mental Status Examination revealed
distressed affect, and somatic preoccupation. Both
the subjects were well oriented, alert, and attentive.
Intelligence, memory, and abstraction were satisfactory
with fair insight and judgment. Psychiatric evaluation
did not reveal any psychiatric comorbidity. The cases
were then referred to the neurologist for an opinion.
The general physical examination was unremarkable.
Medical Journal of Dr. D.Y. Patil Vidyapeeth  ¦  Volume 12  ¦  Issue 4  ¦  July-August 2019
The neurological evaluation showed intact cranial
nerves, coordination, motor and sensory functions.
Ophthalmological examination, including slit‑lamp
biomicroscopy, was normal except for blepharospasm in
case A. Routine investigations and serum calcium level
were within the normal limit. Computed Tomography
Scan and Magnetic Resonance Imaging revealed no
neuroparenchymal abnormality. Case A was diagnosed
with oromandibular dystonia and was given botulinum
toxin type  A injection and clonazepam 0.5  mg on SOS
basis with which patient showed dramatic improvement
within 2 days. This was assessed by the neurologist
based on complaints and clinical examination. On review
after a month, she remained symptom‑free. Case B was
diagnosed with oromandibular dystonia and was started
on tetrabenazine 25 mg/day which was up‑titrated to
50 mg/day in a week and trihexyphenidyl 4 mg/day with
which 30% betterment was reported. She was lost to
follow‑up thereafter. Thus, in both the cases, a regular
and long‑term follow‑up was not possible. Hence, the
sustained benefit with treatment could not be ensured in
these cases.
Discussion
Few of the clinical features of the above‑mentioned cases
were suggestive of dissociative motor disorder such
as movements subsiding, whenever patient engaged in
conversation, temporary disappearance of symptoms with
sleep. However, the symptoms of OMD can be worsened
by emotional factors and can be relieved by sleeping,
relaxing, and talking. The gesteantagoniste  (GA) is a
voluntary procedure that briefly mitigates the severity of
dystonic movements. GA may either be a sensory trick,
in which patients may temporarily reduce dystonia by
gently touching the face or neck, biting the toothpick/lip
or as a forcible trick, like applying counter‑pressure in
opposition to the dystonic head turn, may be necessary
for severe forms of dystonia. The tactile/proprioceptive
sensory gating modulating the sensorimotor integration
and diminishing abnormal dystonic motor output
explains the antidystonic effect of GA. Points against
the diagnosis of dissociative disorder were the late age
of onset, the absence of significant stressor in temporal
relation with the onset, absence of gain and good
premorbid personality. In addition, there was no account
of the comorbid or history of psychiatric disorder. The
diagnosis of OMD is based on history and clinical
examination and there is no gold standard test to confirm
the diagnosis. As a result, it is often misdiagnosed as a
temporomandibular joint syndrome, hemifacial spasm or
psychological disorder, and subsequently, there is a delay
in treatment or patients are managed incorrectly. It is
essential to exclude all secondary causes such as drugs,
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Shailaja, et al.: Oromandibular dystonia
trauma, associated neurological disorders like Wilson’s
disease, etc. A  neurological examination followed by an
MRI must be performed to evaluate for stroke or space
occupying lesion of the basal ganglia. Ceruloplasmin
level and slit‑lamp biomicroscopy to be carried out to rule
out the possibility of Wilson’s disease. Electrocardiogram
and Oromandibular dystonia screening questionnaire
may be of some assistance in the differentiation of
oromandibular dystonia from other conditions.[9‑11]
Oral medication is the first line of treatment. Oral
antidystonic medication includes anticholinergic drugs,
benzodiazepines, and anticonvulsants. However, the
effects are variable. Then, next line of treatment is
injections of botulinum A, though the effects are
temporary, rapid symptom relief and high response rate
of 90%–95% is reported. The GA can be used as an
adjuvant to pharmacotherapy. Local anesthetic blocks,
speech therapy, and the use of oral sensory devices
constitute other therapeutic options for OMD. More
recently, deep brain stimulation of bilateral globus
pallidus interna has emerged as a therapeutic option in
patients with intractable OMD.[3,12‑16] As patients with
OMD face social, emotional, and nutritional problems,
a holistic approach is crucial. Antidepressant medication
and supportive psychotherapy benefit patients suffering
from depression and anxiety. Nutritional issues can be
resolved by the use of texture modified diets, nutritional
supplement drinks, and food fortification.[17]
Declaration of patient consent
The authors certify that they have obtained all
appropriate patient consent forms. In the form the
patient(s) has/have given his/her/their consent for his/
her/their images and other clinical information to be
reported in the journal. The patients understand that their
names and initials will not be published and due efforts
will be made to conceal their identity, but anonymity
cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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