Multidisciplinary management of a partially edentulouspatient with oromandibular dystonia: A clinical report

1. Journal Club Presentation
Multidisciplinary management of a
partially edentulous
patient with oromandibular
dystonia: A clinical report
Neeraja M menon
Ⅱ MDS

2. Content
 Introduction
 Materials and method
 Result
 Discussion
 Related articles
 Summary

3. Introduction
 Oromandibular dystonia (OD) is a focal dystonia whereby
repetitive or sustained spasms of the masticatory, facial, or lingual
muscles result in involuntary, and possibly painful jaw opening,
closing, deflecting, retruding, or a combination of the above.
 It is a progressive condition.
 Although the pathophysiology of dystonia is uncertain, it is
thought to be associated with dysfunction of the basal ganglia
and dystonic movements due to loss of motor control.

4. Etiology
 Primary
Idiopathic,
Inherited or
Familial with genetic predisposition
 Secondary
Peripheral trauma or surgical incidents,
Diseases of the brain (neurodegenerative disorders,
cerebral infarction)
Medications (drug-induced dystonia)

6. Dystonia is classified and defined on 2 axes:
• a. Age of onset
• b. Body distribution
• c. Temporal pattern
• d. Associated features
Axis I
• a. Inherited
• b. Acquired
• c. Idiopathic
Axis II

7. Patients may present with:
► Jaw opening oromandibular dystonia,
► Jaw closing oromandibular dystonia ,
► Jaw deflecting oromandibular dystonia,
► Jaw retruding oromandibular dystonia,
Or a combination of any of these.

8. Epidemiology
Types of Dystonia Total no: of cases F:M ratio
CD 2634 1:5
SD 1411 2:0
BL 739 2:0
UL 296 0:6
OMD 37 3:1
Adapted Movement Disorders Special Issue: Advances
in Dystonia Volume 28, Issue 7, pages 926–943, 15 June
2013

9. Epidemiology
No: of cases %
Primary 11 44
Nuero degenerative diseases (Parkinson disease,
Huntington’s disease, etc)
9 36
Secondary nueroleptic 3 12
Fuctional 12 8

10. Muscles possibly involved
 Masseter
 Temporalis
 Orbicularis oris
 Medial pterygoid
 Lateral pterygoid
 Digastric
 Geniohyoid
 Mylohyoid

11.  The diagnosis of OMD is clinical and based on information
from the individual and the physical and neurological
examination.
 Misdiagnosis commonly includes temporomandibular
disorders (TMD) such as bruxism or spontaneous condylar
dislocation, hemimasticatory or hemifacial spasms and
psychologic manifestations on neurological examination.

12.  Many patients report that they have learned to alleviate the
abnormal movements by “sensory tricks” or “gestes
antagonistes,” such as applying finger pressure beneath the
chin, tongue thrusting, or lip biting.
 Dental devices have been reported to be capable of creating a
sensory trick that may work like these approaches.

13. Treatment
 No curative treatment exists for OMD, but several treatment
strategies may help to reduce the severity of the symptoms.
 Impairment-based approaches can be listed as
♦ pharmacological treatment,
♦ botulinum toxin type A (BTX) injections,
♦ behavioral therapy,
♦ muscle afferent block therapy, and
♦ operative therapy

14. Multidisciplinary management of a
partially edentulous
patient with oromandibular dystonia: A
clinical report
Sakar O, Matur Z, Mumcu Z, Sesen P, Oge E. The Journal of prosthetic dentistry.
2018

15. Aim
This clinical report presents a therapy that combines BTX
injections and the fabrication of a mandibular, customized
occlusal splint and a maxillary, modified removable complete
denture for a partially edentulous patient with OMD.

16. Clinical report
 62 year old female
 c/c: abnormal speech and masticatory muscle pain.
 According to the axis I classification, she had late-adulthood,
focal, progressive, action-specific, and isolated OMD, and
according to the axis II classification, it was an idiopathic form
of OMD.
 Neurological examination revealed a moderate tongue
protrusion with jaw opening that only occurred during speech.

17.  The patient mentioned that her speech improved significantly
while holding a napkin or a lump of sugar in the molar area or
clenching her teeth.

18.  Before her symptoms began.
♦ mandibular incisors and the left canine present.
♦ well-managed mandibular FPD,
♦ a precision attachment-retained RPD
♦ a maxillary CD

19.  The pain in her masseter muscle was thought to be related to
the absence of the closest speaking space because of
continuous clenching.

20.  Before prosthetic intervention,
the bilateral lateral pterygoid (20 units),
digastric (20 units), and
genioglossus muscles (10 units)
were injected with BTX (Botox; Allergan Pharmaceuticals), but
neither significant relief in muscle pain nor any improvement in
speech was achieved.

21. Treatment
 A custom occlusal device with an
increased occlusal vertical dimension
(OVD) that fit over the mandibular RPD
was designed to replicate the patient’s
sensory tricks.
 Shallow, inclined excursive paths were
made so as not to lock the mandible in
its maximal intercuspal position but to
provide freedom in excursive
movements.

22.  The increase in the OVD was adjusted over several sessions
according to feedback from the patient.
 The resulting OVD was described as comfortable by the
patient, and the adjustment started from the maximum height
at which she could comfortably close her lips.

24.  BTX injections were suspended during prosthetic intervention
and restarted after delivery of both the occlusal device and
the modified maxillary denture.

25. A. Right masseter muscle.
B. Left masseter muscle.
C. Genioglossus muscle

26.  The patient was soon able to maintain a comfortable facial
posture, and her speech improved to a clearly understandable
level.
 Quantitative evaluation of her pain scores and speech quality
were made with a visual analog scale (VAS) and Oral Health
Impact Profile 14 (OHIP-14).

27.  According to the VAS scores, her speech quality increased
from 3 to 7, and the pain scores decreased from 5 to 2.
 OHIP-14 scores also improved significantly after treatment.
 After 3 years of follow-up, no occlusal disharmony or
temporomandibular disorder was noted.

28. Discussion
 BTX injections are a primary treatment for OMDs.
 However, secondary resistance may develop in some patients
because of immunogenic causes or the ‘whack-a mole
phenomenon,’ which is that when a dystonic muscle has been
relieved by BTX therapy, one of the other muscles with a
similar function may begin to present similar symptoms

29.  In addition to customized occlusal splints, modification of the
existing dentures should be considered as a straightforward,
non-invasive, and reversible treatment alternative for patients
with OMD who use removable complete or partial dentures.

30. Oral Rehabilitation with Osseointegrated
Implants in a Patient with Oromandibular Dystonia
with Blepharospasm (Brueghel’s Syndrome):
A Patient History
 Peñarrocha M, Sanchis JM, Rambla J, Sánchez M. International
Journal of Oral & Maxillofacial Implants. 2001

31. Aim:
The present article describes a patient with oromandibular
dystonia who was rehabilitated with mandibular overdentures
supported by endosteal implants.

32. CASE REPORT
 67-year-old woman
 2 years since beginning of symptoms
 traumatic ulcerations of the lower lip
 The dystonia was being treated with
clonazepam (6 mg/day) and amitriptyline
(75 mg/day) since 1 year.

34.  The postoperative course was uneventful. Four months later, a
mandibular overdenture supported by 2 endosteal implants,
as well as a conventional complete maxillary denture, were
positioned

35.  The patient reported that the oral rehabilitation improved
mastication and speech.
 Five years after placement of the implants, the latter show
osseointegration with prosthesis stability, and the patient
maintains adequate occlusion.
 Both the surgical phase and the prosthetic rehabilitation
period were uneventful and, in the 5 years of follow-up, the
patient slowly stabilized her oromandibular dystonic
movements, with improved function and esthetic results.

36. Discussion
 Tooth loss is thought to cause a decrease in afferent
proprioceptive impulses and an alteration of the modulation
mechanisms of the central nervous system, which may trigger
dystonia.
 Prosthetic rehabilitation is important in helping to ensure a
normal occlusion that is capable of securing a stable
mandibular position and adequate muscle rest without
worsening the dystonic movements.

37.  In this sense, complete denture rehabilitation poses serious
difficulties, especially in the mandible, because of the marked
instability involved.
 This problem can be resolved by placing mandibular implants
that facilitate adequate prosthetic stability and the restoration
of occlusion.

38. Oromandibular dystonia: A clinical
report
 Schneider R, Hoffman HT. The Journal of prosthetic dentistry.
2011

39. Aim
A multidisciplinary treatment approach is presented including
the fabrication of a dental prosthesis to reduce/eliminate the
symptoms of OMD.

40. Case report
 60 year old female
 Abnormal speech and lisping since 2
years
 Hyperkinetic dysarthria and symptoms
getting worse
 Weight loss and followed a general diet
to help prevent aspiration
 Her speech improved significantly when
holding 2 to 3 tongue blades between
her right posterior dentition

41.  Diagnostic impressions
 Facebow transfer and centric relation record
 A maxillary acrylic resin prosthesis with a pivot on the right
side and retained with ball clasps for the initial trial period.

44.  At the 1 week and 1 month recalls there was complete
resolution of symptoms while she was wearing the prosthesis.
The TMJ remained asymptomatic during this period.

45.  At the 3-month recall the symptoms had returned, but with
less intensity.
 In consultation with the otolaryngologist, the authors
determined that moving the pivot point with the chair-side
addition of acrylic resin would perhaps trigger a positive
neurologic response with a decrease in symptoms.
 Following modification of the prosthesis there was slight
improvement of the symptoms but not complete resolution.

46. Discussion
 Currently, there are no reports in the literature describing the
long term effects of such a device.
 The initial improvement, of unknown duration, has currently
not been explained definitively in the literature.
 It is thought that changes in neuromuscular proprioception
may cause the mechanism that initiates the dystonia to revert
to normal until the nervous system becomes accustomed to
the position and causes the patient to demonstrate the
oromandibular dystonia symptoms again.

47. Use of an oral sensory feedback device in
the
management of jaw-opening dystonia
Verma SP, Sinha UK. Otolaryngology-Head and Neck Surgery.
2009

48.  Jaw-opening dystonia (JOD) is a type of oromandibular
dystonia in which patients suffer from prolonged lateral
pterygoid muscle contraction causing sustained jaw opening.
 Patients have difficulty eating and speaking, often drool
uncontrollably, and suffer from a poor quality of life.
 Therapeutic options for this disease are limited, making this a
challenging problem to address.

49. Aim
This article describes the use of a novel sensory feedback device
in addition to conventional treatment with oral medication and
botulinum toxin A (Botox) injections in the management of JOD.

50. Method
 2 males and 3 females with idiopathic JOD .
 The average age of our patients was 57 years.
 Treatment approach was three tiered.

51. A standard medical regimen was prescribed by the neurologist.
Transoral injections of Botox into the lateral pterygoid muscles.
An oral SFD was delivered to each patient.
Ⅰ
Ⅱ
Ⅲ

52.  The device was clear and custom molded to fit
along the patient’s mandibular teeth.
 A peg of silicone, one cm in height and
spanning three molar teeth provided pressure
to the corresponding maxillary molars.
 Patients were educated to bite down on the
device to overcome the activity of the lateral
pterygoid muscle.
 Application of this device allowed for rapid
relaxation of the affected dystonic muscle
group.

53. Result
 All five patients with JOD treated
with the SFD were able to
immediately overcome symptoms
with intraoral placement of the
device.
 After use of this device, the patients
required Botox injections less
frequently as demonstrated

54. Discussion
 Tactile or sensory stimulation in the region of the affected
muscle group causes relaxation of dystonic muscles. Sensory
tricks are a hallmark of dystonia and are dramatic when
present.
 In the five patients the sensory trick was triggered by
applying light pressure on the molar teeth, which caused
lateral ptyergoid muscle relaxation, allowing patients to
overcome their dystonia and close their mouths

55. Coronoidectomy as treatment for
trismus due to jaw closing
oromandibular dystonia
 Yoshida K. journal of the Movement Disorder Society. 2006

56. Aim
Surgical resection of the bilateral coronoid processes for 2
patients with severe trismus due to jaw-closing dystonia, in
whom treatment by botulinum toxin injection or muscle afferent
block therapy had been ineffective.

57. Case report
 61-year-old female
 Involuntary contraction of jaw-closing muscles that made her
unable to open the mouth fully
 The restricted mouth opening gradually became worse

58.  Her mouth was abruptly closed when attempting to
voluntarily open it because of involuntary jaw-closing muscle
contraction.
 It also was difficult for her to speak clearly and she was only
consuming soft food.
 Jaw hypomobility was temporarily relieved by simply touching
the teeth.

59.  Electromyographic examination

60.  5 to 10 mL of 0.5% lidocaine and 0.5 to 1 mL of 99.5% alcohol
into the bilateral masseter and temporalis muscles.
 The symptoms were slightly relieved, but the effects were
transitory.
 The 20 to 50 units of the Botulinum toxin type A was injected
bilaterally into the masseter and temporal muscle.
 The patients could open their mouths slightly more easily than
before. However, the effects did not last long.

61. Coronoidotomy
 Bilateral coronoidotomy by the intraoral approach was done
under general anesthesia.
 Using a standard intraoral approach, the coronoid process was
cut from the anterior aspect of the ramus to the sigmoid
notch using a fissure bur and chisel.

62. Result
 The inter-incisal jaw opening degree
increased dramatically after
coronoidotomy to 50 mm.
 After surgery, the patients showed
significant improvement in
mastication and were able to
consume any type of food.
 Her speech became normal after
treatment.
 The overall subjective improvement
was 90%

63. Conclusion
 The severe jaw hypomobility in these patients may have
initially resulted from jaw closing muscle contracture due to
oromandibular dystonia and may have been secondarily
intensified by a dense, inelastic tendon and a continuous
contractile force at its point of attachment.
 For such patients, conservative therapies, including muscle
afferent block or botulinum toxin injection, may be ineffective.

64. Summary
 Treatment of OMD is multidisciplinary and dentists should be
aware of the symptoms.
 Treatment strategies are varied and are most effective when
focused on the underlying etiologic factors.