temporomandibular joint dysfunction in marfan syndrome
TRANSCRIPT
Temporomandibular joint dysfunction in Marfan syndrome
Oskar Bauss, Dr med, Dr med dent,a Reza Sadat-Khonsari, Dr med dent,b Christian Fenske, Dr med dent,
PhD,c Werner Engelke, Dr med, PhD,d and Rainer Schwestka-Polly, Dr med dent, PhD,e Hannover,
Germany, Geneva, Switzerland, Gottingen, Germany, Hamburg, Germany, and Minden, GermanyHANNOVER MEDICAL SCHOOL, UNIVERSITY DENTAL SCHOOL OF GENEVA, GEORG-AUGUST UNIVERSITYGOTTINGEN, UNIVERSITY OF HAMBURG, AND GENERAL HOSPITAL MINDEN
Objective. The aim of this study was to examine the prevalence of signs and symptoms of temporomandibular joint (TMJ)dysfunction in persons with Marfan syndrome.Study design. A questionnaire was distributed to 350 patients with Marfan syndrome. Twenty-one patients wereadditionally subjected to a clinical examination and magnetic resonance imaging (MRI) of the TMJ.Results. The prevalence of symptoms of TMJ dysfunction was 51.6% (n = 145), with 24.2 % (n = 68) indicating symptomsof subluxation, and 34.9% (n = 98) of the patients already undergoing medical treatment for their TMJ problems.Anterior disc displacement with and without reduction was observed in 17 of the examined patients (81.0%), with 4 ofthese patients additionally showing osteoarthrosis of the affected temporomandibular joints.Conclusion. TMJ dysfunction appears to be an important aspect in Marfan syndrome.(Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;97:592-8)
Marfan syndrome was first described in 1896 by the
French pediatrician Antoine Bernard-Jean Marfan1 and
represents a connective tissue disorder with dominant
autosomal inheritance. The incidence is estimated to be
at least 1 case per 10 000 individuals.2-4 The connective
tissue dysplasia underlying Marfan syndrome is caused
by a mutation in the fibrillin gene on chromosome 15,
which results in quantitative and/or qualitative changes
of fibrillin; 25% to 35% of all cases are most likely
results of new mutations.5-8 Fibrillin, which represents
an important component in microfibrils and in the elastic
fibers of the connective tissue, occurs in many areas of
the human body (eg, in the skeletal and cardiovascular
systems, and in the eyes).9-11 The diagnostic features of
Marfan syndrome particular to each organ system have
been described elsewhere.12-17
The authors wish to thank the Deutsche Marfan Hilfe e.V. (German
Marfan Foundation) for its support in carrying out this study.aAssistant Professor, Department of Orthodontics, Hannover Medical
School, Germany, and Research Fellow, Department of Orthodontics,
University Dental School of Geneva Geneva, Switzerland.bAssistant Professor, Department of Orthodontics, Georg-August-
University Gottingen, Germany.cAssociate Professor, Department of Prosthodontics, University of
Hamburg, Germany.dProfessor and Head, Department of Oral and Maxillofacial Surgery,
General Hospital Minden, Germany.eProfessor and Head, Department of Orthodontics, Hannover Medical
School, Germany.
Received for publication May 27, 2003; returned for revision Aug 25,
2003; accepted for publication Oct 27, 2003.
1079-2104/$ - see front matter
� 2004 Elsevier Inc. All rights reserved.
doi:10.1016/j.tripleo.2003.10.024
592
In addition, various publications have reported
craniofacial and oral manifestations of patients with
Marfan syndrome.8,18-24 Though joint hypermobility is
a common finding in Marfan syndrome,25-29 only scant
attention was paid in the past to temporomandibular joint
(TMJ) dysfunction.8,22,30 Therefore, the aim of the
present study was to examine the prevalence of sub-
jective TMJ complaints in persons with Marfan syn-
drome and to present the results obtained from the
clinical examination and magnetic resonance imaging
(MRI) of 21 patients. For assessment of the prevalence of
subjective TMJ complaints in persons with Marfan
syndrome, a mail surveyebased research approach was
found appropriate since these cases are few and far apart.
PATIENTS AND METHODS
QuestionnaireIn collaboration with the German Marfan Foundation,
a questionnaire was distributed to all 350 members with
Marfan syndrome; 225 (64.3%) persons were female and
125 (35.7%) were male. The mean age was 41.3 years.
To our knowledge, no previous reports have examined
the prevalence of Marfan syndrome in Germany.
However, since the incidence is estimated to be at least
1 case per 10 000 individuals,2-4 it can be concluded that
the sample size represents about 5% of the number of
affected individuals in Germany.
The objective of the questionnaire was to determine
the frequency of symptoms of functional disorders
involving the TMJ. A standardized questionnaire was
used during this investigation.31 In order to obtain more
detailed information with respect to the prevalence of
TMJ subluxation, the cited questionnaire was modified
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Bauss et al 593
with some additional questions. Hence, the respondents
were also specifically asked about temporary restrictions
to their mouth-opening movement and jolting move-
ments in the TMJ area, as well as restrictions to mouth
closing, as found in TMJ subluxation.32 In addition, in
cases where respondents were undergoing medical
treatment for their TMJ complaints, they were asked
about the type of treatment. Multiple listings of
complaints were possible.
Clinical examination and MRIAll 46 respondents living not more than 125 miles
from the Department of Oral and Maxillofacial Surgery
in Minden were invited for an additional clinical exami-
nation and MRI of the TMJs; 21 patients with Marfan
syndrome responded to this invitation. The average age
of the 11 female and 10 male patients was 38 years
(range, 17-57 years). Thirteen patients (61.9%) reported
no TMJ symptoms and were free of pain, whereas the
remaining 8 patients (38.1%) reported subjective com-
plaints in the TMJ area (TMJ sounds, pain on movement
of the mandible, symptoms of TMJ subluxation).
All subjects were examined clinically prior to MRI
being performed by an experienced investigator. The
clinical examination followed a standardized protocol33
and included evaluation of the mandibular range of
motion (maximum unassisted and maximum assisted
jaw opening, and lateral movements), TMJ palpation and
registration of TMJ pain during unassisted or assisted
mandibular opening, muscle palpation, and joint sound
analysis. The maximum unassisted and maximum
assisted jaw openings were measured as the interincisal
distance plus central incisor vertical overbite, using
a millimeter ruler. The maximum unassisted jaw opening
was measured at maximum opening under voluntary
effort. Assisted maximum mandibular opening was
measured by expanding the active opening while the
patient’s musculature was relaxed. Lateral movements
were registered relative to the maxillary midline, with the
teeth slightly separated. TMJ pain on palpation was
assessed through bilateral manual palpation of the lateral
aspect of the condyle. Pain during unassisted mandibular
opening was registered by asking the patient to open the
jaw as far as possible. The parameter of TMJ pain during
assisted opening was evaluated by applying force to the
lower and upper incisors with the middle finger and
thumb. A positive pain score was recorded by the
examiner if the patient experienced a distinctively pain-
ful sensation in the TMJ during the procedure. Muscle
pain was rated as positive or negative using a bilateral
manual palpation technique on the anterior, middle, and
posterior temporalis, the tendon of the temporalis, and
the superficial and deep masseter. TMJ sounds were
registered by auscultation and manual palpation during
vertical opening and closing movements of the mandi-
ble. If there was uncertainty about the side concerned, the
patient was consulted. Joint sounds were described as
single or reciprocal clicks.34
After the clinical examination, all 21 patients un-
derwent bilateral MRI of their TMJs. MRI was
performed at 1.5T (Magnetom Vision, Siemens AG,
Erlangen, Germany) with a special surface coil (TMJ
coil). Paracoronal and parasagittal slices of each TMJ
were obtained by using an optimized proton-weighted
TSE sequence (TR 2800 ms, TE 15 ms) and a turbo
inversion recovery magnitude sequence (TR 4000 ms,
TE 30 ms, TI 150 ms). A slice thickness of 3 mm and
a 256-matrix with a field of view of 145 mm were used.
Sequential bilateral T1-weighted and T2-weighted
images were made at the closed-mouth and the re-
spective maximum open-mouth positions. For fixation of
mouth opening during the scans, a standardized device
was inserted between the arches in all patients. MR
images were corrected to the horizontal angulation of the
long axis of the condyle. The radiological evaluation was
performed by an experienced examiner who was blind to
the medical history and the clinical diagnosis of the
patient. Those MRI images were selected for analysis of
the disk-condyle relationship (DCR) that depicted the
disk, condyle, articular eminence, and glenoid fossa.
Normal disk position was defined by location of the
posterior band of the disk at the superior or 12 o’clock
position relative to the condyle. Disk displacement was
defined as the posterior band of the disk being in an
anterior, anteromedial, anterolateral, medial, or lateral
position relative to the superior part of the condyle.34
Diagnosis of TMJ DCR was categorized as normal, disk
displacement with reduction, or disk displacement
without reduction. Normal function was registered when
a disk in the superior position in the closed mouth
position maintained a position interposed between the
condyle and the articular eminence in the open mouth
position. Disk displacement with reduction was noted
when a displaced disk in the closed mouth position
assumed a position interposed between the condyle and
the articular eminence in the open mouth position. Disk
displacement without reduction was noted when a dis-
placed disk in the closed mouth position did not achieve
a position between the condyle and the articular
eminence in the open mouth position.35 An MRI
diagnosis of osteoarthrosis was defined by the presence
of flattening associated with subchondral sclerosis,
surface irregularities and erosion of the condyle, or
presence of condylar deformities associated with
flattening, subchondral sclerosis, surface irregularities,
erosion, and osteophytes.36,37
Reliability scores were determined by a blind test-
retest method on 30 consecutive patients (clinical
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGYMay 2004
594 Bauss et al
Fig 1. Frequency of different symptoms of temporomandibular joint (TMJ) dysfunction. 1: Pain at rest; 2: Pain on mouth opening; 3:
Pain on chewing; 4: Joint sounds (clicking or crepitation); 5: Symptoms of TMJ subluxation; 6: Recurring symptoms of TMJ
subluxation.
diagnoses) and on a set of 80 images in 40 randomly
selected subjects (radiological diagnoses), thereby
allowing for intraobserver comparison. Evaluation of
the statistical significance of the diagnostic percentage
agreement between the interpretations was performed
using the k statistical test.38 The intraobserver agreement
for the clinical diagnoses was substantial for pain during
muscle palpation (k = 0.73) and very good for the
remaining clinical diagnoses (k > 0.81). All radiological
diagnoses revealed very good intraobserver reliability
(k > 0.81).
RESULTS
QuestionnaireA total of 281 questionnaires (80.3%) were evaluated;
184 of the respondents were female (65.5%) and 82 were
male (29.2%). The average age was 40.7 years. Fifteen
respondents (5.3%) gave no information concerning
their age and gender.
Subjective complaints (pain, sounds) in the TMJ area
were reported by 145 respondents (51.6%). Fifty-eight
(20.6%) respondents reported pain at rest, whereas 103
(36.7%) reported pain during mouth opening and 56
(19.9%) during chewing. Ninety-two (32.7%) persons
reported subjective TMJ sounds (clicking, crepitation).
Sixty-eight (24.2%) had experienced symptoms of TMJ
subluxation with restricted mouth closing at least once,
and 18 (6.4%) reported recurring subluxation symptoms
(Fig 1). Ninety-eight (34.9 %) respondents reported
having undergone medical treatment for their TMJ com-
plaints in the past. In 86 (87.8%) of these respondents,
treatment had involved interocclusal stabilization ap-
pliances, whereas 12 (12.2%) had undergone surgery.
Clinical examination and MRI findingsSeventeen of the 21 (81.0%) examined patients (9
women and 8 men) showed signs of TMJ dysfunction in
the clinical examination. Four patients (19.0%) revealed
no signs of TMJ dysfunction and a normal position of the
disc in the MRI.
In 9 of the 13 anamnestically pain-free patients,
reciprocal clicking in one (n = 7) or both (n = 2)
temporomandibular joints accompanied by deviation
during opening movement was observed in addition to
some palpatory tenderness of the masticatory muscles.
The quality and occurrence of noise during mandibular
movement could be influenced through superior joint
loading by applying mild pressure at the inferior border
of the mandible toward the TMJ. Four of the 9 patients
experienced discomfort in the affected temporomandib-
ular joints when the joints were laterally palpated. In all
9 patients, the MRI scans showed unilateral (n = 7)
or bilateral (n = 2) anterior disc displacement with
reduction.
Four patients complained of pain in the preauricular
area, which was exacerbated by applying lateral pressure
to the temporomandibular joints. These patients showed
markedly limited mouth opening with deflection of the
lower jaw to the affected side. During the recording of
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Bauss et al 595
Fig 2. Bilateral anterior disk displacement without reduction and disk deformation in a 36-year-old woman with Marfan syndrome.
A, Closed mouth. B, Open mouth. C = condyle; D = disk.
their medical history, all 4 patients reported regular
clicking in the affected joints, which disappeared when
the restricted movement appeared. In all 4 patients, MRI
confirmed the clinical diagnosis of unilateral (n = 3) and
bilateral (n = 1) disc displacement without reduction
(Fig 2).
Four patients (28-36 years) reported bilateral pain
localized to the TMJ. They also showed marked
crepitation during the opening and closing cycle, and
a great discrepancy between unassisted maximum and
assisted maximum opening, which indicated an in-
hibition of maximum opening through muscle activity.
Here, too, pain and tenderness were observed on
palpation of the masticatory muscles. MRI confirmed
the clinical indications for osteoarthrosis. All 4 cases
showed flattening of the condylar surface with osteo-
phyte formation, as well as bilateral anterior disc dis-
placement without reduction (Fig 3).
DISCUSSIONIn contrast to Ehlers-Danlos syndrome, where a pos-
sible effect of the connective tissue dysplasia on the TMJ
has been described in numerous studies,39-45 there are
only a few isolated reports with regard to Marfan
syndrome.8,22,30
The prevalence of subjective TMJ complaints in
Marfan syndrome was examined by Westling et al8 in 76
patients, but without the information being differentiated
as to type of complaint and kind of treatment. Of the
respondents in that study, 56% indicated dysfunctions
and/or pain in the TMJ area and 25% were undergoing
medical treatment for these symptoms; these figures are
comparable with the findings of the present study.
However, previous examinations have determined a high
prevalence of symptoms of TMJ dysfunction in the
population. In a survey conducted in Canada, for
instance, 48.8% of 1002 respondents gave at least 1
positive answer to the 9 questions regarding dysfunction
symptoms, whereas 13% of respondents even indicated
that they experienced pain in action or at rest.46 In
numerous cross-sectional epidemiological studies, the
frequency of symptoms is given as between 12% and
59%.47 An investigation conducted in a normal German
population found a frequency of subjective TMJ
complaints of about 35%.48 Therefore, the results of
the present investigation with respect to the frequency of
ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGYMay 2004
596 Bauss et al
Fig 3. First signs (left side) and advanced stage (right side) of osteoarthrosis of the TMJ with anterior disk displacement without
reduction in a 28-year-old man with Marfan syndrome. A, Closed mouth. B, Open mouth. C = condyle; D = disk.
pain and sounds in the TMJ area in Marfan patients
should not be overrated.
In contrast to the frequency of the above-mentioned
symptoms of TMJ dysfunction, the present study points
to a high prevalence of symptoms of TMJ subluxation in
Marfan syndrome patients. Nearly 25% of the
respondents experienced symptoms of TMJ subluxation,
which is far above the frequency reported in previous
studies in a normal population (2%-3%),49 and might be
explained by hypermobility of the disc and the TMJs,
caused by the connective tissue disorder in Marfan
syndrome.
More than 10% of the respondents in the present study
had undergone surgery for their TMJ complaints. As
opposed to Ehlers-Danlos syndrome,40-42,44 there have
to our knowledge been no previous accounts of surgical
management of TMJ disorders in Marfan patients.
Previous reports on the surgical treatment of TMJ
dysfunction in Ehlers-Danlos syndrome have stated that
the conventional treatment of discal plication should be
used with caution due to the connective tissue changes
occurring in this disease.40,42 This might also apply to
the surgical treatment of TMJ disorders in Marfan
syndrome. However, no further information was avail-
able on the kind and success of surgical treatment
performed.
As far as we know, no previous investigation with
a larger sample size has examined the prevalence of signs
of TMJ dysfunction in Marfan syndrome. Barr30
reported osteoarthrosis of the TMJ in a Marfan patient
before the age of 30, as well as bilateral TMJ clicking and
capsular tenderness in a few other patients. Nally22
observed unilateral TMJ subluxation in 1 case with
Marfan syndrome. These reports correspond to the
findings of the present investigation; 81% of the
examined patients showed unilateral or bilateral anterior
disc displacement. As the frequency in asymptomatic
volunteers has been reported to vary between 20% and
30%,50,51 a higher frequency of anterior disc displace-
ment in patients with Marfan syndrome might be
concluded. Thus, as in Ehlers-Danlos syndrome, con-
nective tissue changes in Marfan syndrome also appear
to lead to hypermobility of the disc and the TMJs.
However, there are certain aspects that have to be
considered when interpreting the results of the present
investigation. Subjects with Marfan syndrome are very
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Bauss et al 597
rare and only 21 of the invited 46 patients living near the
clinic where the examination was performed responded
to an invitation. Therefore, the number of examined
patients was lower than in other studies dealing with
temporomandibular disorders. In addition, the present
study was performed without a matched control group of
patients without Marfan syndrome so that the results
could only be compared to the findings of a previously
published investigation in a normal German popu-
lation.48
However, the results of the present study suggest that
TMJ dysfunction might represent an important aspect in
Marfan syndrome. They also support the theory that, in
addition to occlusal, psychological, and parafunctional
factors,52-56 constitutional factors such as general dis-
orders of connective tissue57-59 also represent an im-
portant factor in the emergence of TMJ dysfunction.
CONCLUSIONSAn increased prevalence of TMJ subluxation and
signs and symptoms of TMJ dysfunction may be ex-
pected in patients with Marfan syndrome. This supports
the theory that connective tissue disorders should be seen
as an important etiological factor in the emergence of
TMJ dysfunction.
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Reprint requests:
Oskar Bauss, Dr med, Dr med dent
Department of Orthodontics
Hannover Medical School
Carl-Neuberg-Strasse 1
30625 Hannover, Germany