Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP)Temporomandibular disorders are a set of musculoskeletal dysfunctions within the masticatory system, with multiple etiologies. Objective: Since craniocervical dystonia can involye the same neuromuscular structure as the temporomandibular joint, we sought to assess the correlation between temporomandibular disorders and craniocervical dystonia. Method: We applied the Research Diagnostic Criteria for Temporomandibular Disorders to 42 patients with craniocervical dystonia, in order to identify orofacial pain and temporomandibular characteristics on the day of botulinum toxin injection. Results: Twenty-two patients (52.3%) reported temporomandibular joint pain; 24 (57.1%), joint sounds; 20 (47.6%), masticatory muscle pain; and 21(50%), diminished jaw mobility. The patients with oromandibular dystonia presented temporomandibular disorders more frequently than did patients with other types of craniocervical dystonia (p<0.001). Conclusion: Temporomandibular disorders occur frequently in patients with oromandibular dystonia. Further studies should address the proper treatment of temporomandibular disorders associated with dystonia. Key words: botulinum toxin, cervical dystonia, cranial dystonia, temporomandibular disorders.696896899Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP)Boehringer IngelheimRocheIpsenFundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP

Campos, LS

Costa, AL

D'Abreu, A

Franca, MC

Arquivos de Neuro-Psiquiatria

Repositorio da Producao Cientifica e Intelectual da Unicamp

Arq Neuropsiquiatr 2011;69(6):896-899
896
Article
Temporomandibular disorders in 
patients with craniocervical dystonia
André L. Costa1, Lidiane S. Campos2, 
Marcondes C. França Jr.1,2, Anelyssa D’Abreu1,2
ABSTRACT
Temporomandibular disorders are a set of musculoskeletal dysfunctions within the 
masticatory system, with multiple etiologies. Objective: Since craniocervical dystonia can 
involve the same neuromuscular structure as the temporomandibular joint, we sought 
to assess the correlation between temporomandibular disorders and craniocervical 
dystonia. Method: We applied the Research Diagnostic Criteria for Temporomandibular 
Disorders to 42 patients with craniocervical dystonia, in order to identify orofacial 
pain and temporomandibular characteristics on the day of botulinum toxin injection. 
Results: Twenty-two patients (52.3%) reported temporomandibular joint pain; 24 
(57.1%), joint sounds; 20 (47.6%), masticatory muscle pain; and 21 (50%), diminished 
jaw mobility. The patients with oromandibular dystonia presented temporomandibular 
disorders more frequently than did patients with other types of craniocervical dystonia 
(p<0.001). Conclusion: Temporomandibular disorders occur frequently in patients 
with oromandibular dystonia. Further studies should address the proper treatment of 
temporomandibular disorders associated with dystonia.
Key words: botulinum toxin, cervical dystonia, cranial dystonia, temporomandibular 
disorders. 
Disfunção temporomandibular em pacientes com distonia craniocervical
RESUMO
As disfunções temporomandibulares são um conjunto de alterações musculoesqueléticas 
no sistema mastigatório de etiologia multifatorial. Objetivo: A distonia craniocervical 
pode envolver as mesmas estruturas neuromusculares da articulação temporomandibular. 
Nosso objetivo foi avaliar a correlação entre distúrbios temporomandibulares e distonia 
craniocervical. Método: Aplicamos o Critério Diagnóstico para Pesquisa em Disfunção 
Temporomandibular em 42 pacientes com distonia craniocervical a fim de identificar 
dor orofacial e características da articulação temporomandibular no dia da injeção de 
toxina botulínica. Resultados: Vinte e dois pacientes (52,3%) relataram dor na articulação 
temporomandibular, enquanto 24 apresentaram sons articulares (57,1%), 20 dor na 
musculatura mastigatória (47,6%) e redução da mobilidade mandibular foi observada em 
21 pacientes (50%). Os pacientes com distonia oromandibular apresentaram disfunção 
temporomandibular em maior frequência do que aqueles com outros tipos de distonia 
craniocervical (p<0,001). Conclusão: A disfunção temporomandibular é frequente em 
pacientes com distonia oromandibular. Novos estudos devem abordar o tratamento 
adequado das disfunções temporomandibulares associado à distonia.
Palavras-Chave: toxina botulínica, distonia cervical, distonia cranial, disfunção 
temporomandibular.
Correspondence
Anelyssa D’Abreu 
Department of Neurology / UNICAMP
Rua Tessália Vieira de Camargo 126
Cidade Universitária Zeferino Vaz
13083-970 Campinas SP - Brasil
E-mail: anelyssa@hotmail.com
Conflicts of interest
The authors report no 
conflicts of interest
Support
FAPESP
Full financial disclosures of all 
authors for the past year
André L. Costa: none; Lidiane S. 
Campos: travel grant from Boehringer 
Ingelheim and Roche; Marcondes C. 
França Jr: travel grant from Ipsen; 
Anelyssa D’Abreu: travel grant from 
Boehringer Ingelheim and Roche
Received 24 May 2011
Received in final form 5 July 2011
Accepted 12 July 2011 1Neuroimaging Laboratory; 2Department of Neurology, State University of Campinas (UNICAMP), Campinas SP, Brazil.
Craniocervical dystonia is defined as 
a group of neurological disorders char-
acterized by involuntary muscle con-
tractions that frequently cause twisting, 
repetitive movements or abnormal pos-
tures affecting the periocular, perioral and 
Arq Neuropsiquiatr 2011;69(6)
 897
TMD: craniocervical dystonia
Costa et al.
laryngeal musculature, as well as mastication muscles 
and cervical muscles1. Clinically, craniocervical dystonia 
manifests as isolated blepharospasm, laryngeal dystonia, 
cervical dystonia (CD), oromandibular dystonia (OD) or 
any combination of these. 
Temporomandibular disorder (TMD) is a collective 
term embracing a number of clinical problems that in-
volve the masticatory musculature, the temporoman-
dibular joint (TMJ) and associated structures, or both2. 
These disorders are characterized by facial pain in the 
region of the TMJ and the mastication muscles, limita-
tion or deviation in the mandibular range of motion, or 
TMJ sounds during jaw movement and function3. Their 
prevalence varies, depending on whether single or com-
bination symptoms are used to establish the diagnosis of 
TMD4. The prevalence of TMD-associated pain in the 
American population is 4.6% (6.3% for women and 2.8% 
for men), with higher prevalences in the non-Hispanic 
population at younger ages, and increasing prevalence 
with older age5. In Brazil, a validated telephone survey 
including 1,230 inhabitants found that at least one TMD 
symptom was reported by 39.2% of the sample, two symp-
toms by 17.6% and three or more symptoms by 9.2%6. 
There are a close anatomofunctional relationship be-
tween the masticatory system, the cervical region and 
the TMJ. Although associations between cranial and cer-
vical dystonia and the TMD have not been previously in-
vestigated, indirect evidence has suggested that there is 
an association between the two syndromes7.
The aim of this study was to estimate the prevalence 
of TMD in patients with craniocervical dystonia. 
METHOD
Study population
This study was approved by the Internal Review 
Board of our institution, and all patients signed an in-
formed consent form. Thus, the study was conducted in 
accordance with the ethical standards laid down in the 
1964 Declaration of Helsinki. We included 42 individuals 
with craniocervical dystonia (26 females) through reg-
ular follow-up visits to the Movement Disorders Clinic 
at the University Hospital at UNICAMP. The patients’ 
mean age was 56.1 years (range: 28-86; SD=16.8) and the 
mean duration of the disease was 15.1 years (range: 3-49; 
SD=9.4). The patients’ diagnoses of dystonia had been es-
tablished by a movement disorder specialist, and all of 
them were under treatment with botulinum toxin injec-
tions. Five patients presented secondary dystonia (four 
due to neuroleptics and one due to encephalitis), and all 
the others had primary dystonia. We did not include any 
patients with isolated blepharospasm or isolated laryn-
geal dystonia in this evaluation. The patients’ diagnoses 
and dystonia location are presented in Table.
Clinical evaluation
All the patients underwent a standardized protocol 
(The Research Diagnostic Criteria for TMD, RDC/TMD, 
Axis I)8, applied by a trained dentist who was a specialist 
in TMD. The protocol consisted of an interview and sys-
tematic evaluation of mandibular range of motion, joint 
sounds, joint and muscle pain on palpation, and pain on 
unassisted and assisted mandibular opening. 
To calculate the patients’ mandibular range of mo-
Table. Classification of the dystonia findings according to etiology and location.
Etiology Location Absolute number %
Primary Cervical 16 38.05
Cervical/blepharospasm 1 2.4
Cervical/upper arm 1 2.4
Oromandibular 3 7.15
Oromandibular/cervical 3 7.15
Oromandibular/blepharospasm 7 16.6
Oromandibular/cervical/blepharospasm 3 7.15
Generalized (with oromandibular involvement) 3 7.15
Total 37 88.05
Secondary Cervical 3 7.15
Cervical/upper arm 1 2.4
Generalized (with oromandibular involvement) 1 2.4
Total 5 11.95
Arq Neuropsiquiatr 2011;69(6)
898
TMD: craniocervical dystonia
Costa et al.
tion, we measured the maximum opening from a cen-
tral maxillary incisor to the opposing mandibular in-
cisor. We then measured lateral movements relative to 
the maxillary midline with the teeth slightly separated. 
Jaw mobility was rated as normal when the maximum 
interincisal opening was ≥40 mm and the maximum lat-
erotrusive movement was ≥7 mm. When at least one of 
these measurements was lower than the normal values, 
it was defined as impaired. 
We evaluated patients’ TMJ pain on palpation 
through bilateral manual palpation of the lateral aspect 
of the condyle. TMJ pain during unassisted mandibular 
opening was assessed by asking patients to perform max-
imum voluntary jaw opening. We performed assisted 
mandibular opening by applying force to each patient’s 
lower and upper incisors with the middle fingers and 
thumbs. Muscle pain was assessed by bilateral manual 
palpation of the anterior, posterior and middle tempo-
ralis muscle; the tendon of temporalis; the superficial and 
deep masseters; the lateral pterygoid muscle; the anterior 
and posterior digastric muscles; and the cervical muscles.
We evaluated bruxism by means of a standardized 
interview. The patients were asked about grinding or 
clenching of the teeth, and they were examined intra-
orally to revealed any incisal and/or occlusal wear on 
the teeth.
We did not address secondary dental problems 
caused by TMD, in this evaluation. The patients were 
evaluated on the day scheduled for botulinum toxin ap-
plication, prior to injection.
Statistical analysis
We used the Systat 9.0 for Windows software package 
(SPSS, Inc., Chicago, Illinois, USA). We calculated the 
mean, range and standard deviation to describe contin-
uous variables. Comparisons between groups were done 
by means of the t-test and Fisher’s exact test. The signifi-
cance level was established at p<0.05.
RESULTS
Twenty-two patients (52.3%) reported TMJ pain, 
while 24 (57.1%) had joint sounds, 20 (47.6%) reported 
masticatory muscle pain and 21 (50%) presented dimin-
ished jaw mobility. The TMD diagnosis based on RDC/
TMD was: disc displacement (n=23), myofacial pain 
(n=20), arthralgia (n=23) and arthrosis (n=1).
We performed a secondary analysis in which we di-
vided the patients into two different groups: patients 
with oromandibular dystonia (OD) (n=20), and those 
without oromandibular dystonia (NOD) (n=22). There 
were no significant differences in age (p=0.35), disease 
duration (p=0.37) or gender distribution (p=0.23). 
We diagnosed TMD in all OD patients and in three 
NOD patients. We also found bruxism in eight OD pa-
tients and two NOD patients. The mean mouth opening 
was 42 mm for NOD and 39 mm for OD (p=0.4). How-
ever, taking 40 mm to be the normal magnitude for 
maximum mouth opening, only six OD patients had a 
normal mouth opening. Three OD patients had a per-
manent jaw opening.
We found a statistically significant difference be-
tween the two groups regarding the frequency of TMJ 
pain. Seventeen OD patients suffered TMJ pain, while 
only five patients in the NOD groups reported TMJ pain 
(p<0.001). We also found similar differences in all other 
variables studied: presence of joint sounds (p<0.001), 
pain on palpation of the masticatory muscles (p<0.001), 
restriction of movement (p<0.001) and disc displacement 
(p<0.001). For further details see Figure.
DISCUSSION
To our knowledge, this is the first comprehensive 
clinical study on temporomandibular disorders in pa-
tients with craniocervical dystonia. We found a very high 
prevalence of TMD in patients with craniocervical dys-
tonia, especially in those with OD.
The joint disc of the TMJ is vulnerable to injury be-
cause of its limited ability to redistribute the pressure and 
stress within the joint. This makes the TMJ highly sus-
ceptible to overload damage9. OD is probably an impor-
tant precipitating factor in the development of the hyper-
trophic responses in the subchondral bone and disc that 
eventually alter the articular space. Disc displacement is 
believed to result from trauma, due to impact or hyper-
extension of the joint10. Conversely, OD can originate 
from dysfunction at the central and peripheral levels of 
the motor system, and disruption of the sensory pathway 
at the level of the basal ganglia has been implicated11.
Primary pain in one of the structures of the stomato-
gnathic system may also lead to secondary changes at 
Patients with TMD symptoms/signs according to group
5
17
TMJ
pain
Joint
sounds
Muscle
pain
Limitation
movement
Disc
displacement
17 17
8
3 4
7
16
NOD
16
OD
NOD: no oromandibular dystonia; OD: oromandibular dystonia; TMD: temporo-
mandibular disorders.
Figure. Absolute number of patients with TMD symptoms and 
signs according to group.
Arq Neuropsiquiatr 2011;69(6)
 899
TMD: craniocervical dystonia
Costa et al.
distant sites and this may turn into a vicious cycle that 
perpetuates pain12. TMD signs and symptoms were not 
as frequent in CD subjects. Although some studies have 
suggested that there may be an association between head 
and cervical pain and TMD13, our results do not corrob-
orate such findings.
The treatment for craniocervical dystonia relies 
mainly on botulinum toxin injections14. Interestingly, 
some authors have also suggested that botulinum toxin 
injections should be used to treat TMD15. Therefore, 
further studies should now address the impact of reg-
ular botulinum toxin injections on OD-associated TMD, 
including TMJ and masticatory muscle pain, joint 
sounds, disc displacement and movement limitation. 
When only partial improvement in TMD is achieved, 
evaluation and treatment by a TMD specialist may pro-
vide additional benefit.
ACKNOWLEDGEMENTS – The authors are grateful to Dr. Joseph 
Friedman for reviewing this manuscript. 
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Temporomandibular disorders in patients with craniocervical dystonia

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Temporomandibular disorders in patients with craniocervical dystonia

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