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Accepted Manuscript

Atypical Temporomandibular Joint Pain: A Case Report

Charles G. Widmer, D.D.S., M.S. Courtney J. Wold, D.M.D., M.D. Ethan M. Stoll,
M.S., D.O. M. Franklin Dolwick, D.M.D., Ph.D.

PII: S2212-4403(14)01237-1
DOI: 10.1016/j.oooo.2014.08.013
Reference: OOOO 992

To appear in: Oral Surgery, Oral Medicine, Oral Pathology and Oral
Radiology

Received Date: 15 July 2014


Revised Date: 6 August 2014
Accepted Date: 11 August 2014

Please cite this article as: Widmer CG, Wold CJ, Stoll EM, Dolwick MF, Atypical Temporomandibular
Joint Pain: A Case Report, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology (2014), doi:
10.1016/j.oooo.2014.08.013.

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Manuscript Title: Atypical Temporomandibular Joint Pain: A Case Report

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Authors: Charles G. Widmer, D.D.S., M.S.1, Courtney J. Wold, D.M.D., M.D.2 , Ethan M. Stoll,
M.S., D.O. 3 and M. Franklin Dolwick, D.M.D., Ph.D.4

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Associate Professor, Department of Orthodontics, College of Dentistry, University of Florida

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Resident, Department of Oral and Maxillofacial Surgery, College of Dentistry, University of
Florida
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Surgical Pathology Fellow, Department of Pathology, Immunology & Laboratory Medicine,

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College of Medicine, University of Florida
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Professor and Chair, Parker E. Mahan Facial Pain Endowed Professor, Department of Oral and
Maxillofacial Surgery, College of Dentistry, University of Florida
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Address correspondence to:


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Charles G. Widmer, D.D.S., M.S.


Department of Orthodontics
Box 100444, JHMHSC
College of Dentistry
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University of Florida
Gainesville, FL 32610-0444
Phone: 352-273-5696
Fax: 352-846-0459
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e-mail: widmer@dental.ufl.edu
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Disclosures: All authors report no disclosures associated with this case report.

Abstract word count: 84


Complete manuscript word count: 1926
Number of references: 16
Number of figures: 4
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Abstract

Atypical temporomandibular joint (TMJ) pain can consist of an unusual intensity, location

or set of pain descriptors that do not match what is traditionally observed for TMJ capsular pain,

disc displacements or arthritic conditions. An atypical pain report regarding a unilateral TMJ

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pain as the chief complaint is presented in this case report. An overview of typical vs atypical

TMJ pain is also reviewed to highlight unusual signs and symptoms so that the clinician can

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identify these atypical presentations and pursue further diagnostic approaches.

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Pain in the temporomandibular joint (TMJ) typically presents as a low to moderate level of pain

that is enhanced with function such as chewing. When a patient presents with an atypical, high

level of pain with mandibular function, a further comprehensive evaluation of the

temporomandibular joint is indicated. The following case report provides details of one patient

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with an unusually high level of TMJ pain only at maximum opening and atypical pain descriptors

associated with his pain.

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CASE REPORT

A 69 year old African-American male presented with a chief complaint of left-sided jaw

pain and a history of pain in his left temporomandibular joint (TMJ) area only while chewing and

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opening wide during yawning. This pain started approximately five years prior to being
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examined and had increased over the last three years. Based on the McGill Pain

Questionnaire, he described his pain as sharp, shooting, hurting and aching pain with other
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descriptors including blinding, troublesome, piercing, tight and dreadful. His highest level of
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pain was rated at an intensity of 90/100 on a Visual Analog Scale (VAS) and was only elicited
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when he was opening his jaw wide during yawning. A positive history of reciprocal clicking in

the left temporomandibular joint was reported during mandibular opening along with crepitus (a
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grinding sound). He denied a history of facial trauma but acknowledged diurnal bruxism

(clenching his teeth). His medical history was positive for diabetes, hypertension and arthritis.
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Clinical examination found no masticatory or cervical muscles tender to palpation, no


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lymphadenopathy and no palpable pain lateral to the temporomandibular joints or while loading

either TMJ. Auscultation of the TMJs revealed course crepitus near his maximum opening of 45

mm. Maximal protrusive and right lateral excursion elicited pain in his left TMJ that duplicated

his chief complaint and no pain was elicited with left lateral excursion. A recent panoramic film

depicted the left condyle with an intact cortical lining but had a vague radiolucent region in the

center of the condyle that was not well-defined (Fig. 1).


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Impression

The differential diagnosis for this clinical presentation included: (1) osteoarthritis

with a subchondral cyst; (2) a tumor involving the mandibular condyle; or (3) an

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aneurysmal bone cyst. Given the patient’s age and sex, the most likely tumor causing

this lesion would be from a metastatic lung carcinoma or prostate adenocarcinoma1.

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Metastatic lesions in the mandible usually present as radiolucent defects. These

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lesions may be well circumscribed similar to this patient’s lesion; however, more often

they are irregular with a “moth-eaten” appearance2. Additionally, the prolonged duration

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of this patient’s lesion is inconsistent with the rapid growth typically seen in malignant
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lesions.

Although the cause and pathogenesis of aneurysmal bone cysts is poorly


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understood, it is an intra-osseous collection of blood-filled spaces that has a fibrous


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connective tissue lining. Pain and swelling are common; however, crepitus is rare.
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Aneurysmal bone cysts typically affect patients under the age of 303, which places this

condition lower on the differential diagnosis for this patient.


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The age of the patient and the presence of distinct crepitus within the TMJ would

suggest that the most commonly encountered diagnosis of osteoarthritis may be a


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contributing factor for his pain4. The reciprocal clicking was consistent with an anteriorly
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displaced disc with perforation of the retrodiscal attachments to allow bone-to-bone

contact at maximal opening and was responsible for the crepitus sound. However, the

unusual presentation of pain that is experienced only at maximal opening or maximal

protrusion and not associated with loading the TMJ is not typical of an active

inflammatory process involving bony remodeling of articular tissue. The high level of
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pain reported by the patient was also a concern, although the pain was not constant (a

common characteristic of tumors). Atypical pain presentations are indications for further

hard or soft tissue examination of the patient.

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Additional Diagnostic Testing. A cone beam CT (CBCT) study was acquired to allow a more

detailed examination of the left TMJ (Fig. 2). The CBCT images depicted a 0.8 x 0.7 x 0.9 cm

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lytic lesion that occupied approximately the lateral half of the left mandibular condyle with loss of

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cortical integrity on the superior-lateral articulating surface. To further explore the status of the

bony remodeling and to explore the potential for an active infection, further diagnostic testing

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was pursued using skeletal scintigraphy with correlative gallium imaging. An increase in
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Technetium-99 was observed in the left TMJ condyle (Fig. 3) suggesting an increase in

metabolic activity but gallium imaging was negative supporting the lack of infection. The results
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of the scintigraphy study were consistent with a reactive process. The patient was subsequently

scheduled for a biopsy of the cyst within the left mandibular condyle. During entry into the left
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temporomandibular joint space, an abundant amount of synovial fluid was encountered with no
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defined articular disc. The cystic cavity identified in the condyle from the CBCT was exposed

from a posterior approach and it was found that the cavity extended to the most superior aspect
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of the condylar head perforating the articular surface. The entire contents of the cystic cavity

were removed and submitted for pathological examination and interpretation. The results of the
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biopsy were consistent with dense fibrocollagenous tissue with fragments of non-viable
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cartilage, secondary to osteoarthritis (Fig. 4). The patient has been followed for six years

following his surgery and has not had a return of pain. Since there has been no TMJ pain or

dysfunction, no follow-up radiographs were available to evaluate his current temporomandibular

joint bony anatomy. However, he continued to have crepitus in the left temporomandibular joint.
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Osteoarthritis Involving the Temporomandibular Joint.

Osteoarthritis (OA) is defined by the Osteoarthritis Research Society International

(OARSI) as “a progressive disease of synovial joints that represents failed repair of joint

damage that results from stresses that may be initiated by an abnormality in any of the synovial

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joint tissues, including articular cartilage, subchondral bone, ligaments, menisci (when present),

peri-articular muscles, peripheral nerves or synovium”5. TMJ osteoarthritis (OA) has been

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described as a four-stage pathogenesis: (1) initial and repair; (2) early; (3) intermediate; and (4)

late6. Pain is reported in the three later stages, crepitus is palpated in the last two stages while

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subchondral cysts can be observed in the last stage. Sex differences for the occurrence of

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degenerative changes in the temporomandibular joint have been equivocal. One study of 22

individuals (age 60-89) at autopsy identified more degenerative temporomandibular joints in


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females7 while another report investigating 259 human skulls (age range 18-100) supported a

higher overall prevalence in males8. A multicenter study of 204 consecutive adult patients (age
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range 18-90) with pain, TMJ sounds or limitation of mandibular opening was evaluated for
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degenerative changes in the TMJ and, based on logistic regression, found that age and sex
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(females) were predictors of degenerative changes with age as the most dominant predictor9.

However, this study was not a cross-sectional epidemiological study and had a predominant
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female representation (75%). Thus, it remains unclear whether sex has a predominant effect on

the development of TMJ osteoarthritis whereas older age has a much higher relationship.
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The presentation in this reported case is consistent with late stage OA. Pain was elicited
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when the superior aspect of the condyle (the location of the subchondral cyst) was

approximated to the opposing bone of the articular eminence such as when the mandible was

protruded or when he opened maximally. Since the anterior-superior surface of the condyle

was unaffected and this is where a load is exerted when biting in centric occlusion, direct

pressure was not induced on the articular tissue that was degenerated and there was no biting

pain reported during the clinical examination.


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Clinical Factors Affecting Interpretation

Common intracapsular conditions that can cause pain in the temporomandibular joint

include disc displacement with or without reduction, arthridities and post-traumatic injuries.

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Using the report of pain in combination with joint and muscle palpation, joint loading, range of

motion studies and joint sounds, a diagnosis of an intracapsular source of pain can be achieved

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without the need for advanced diagnostic techniques such as imaging. Most of the patients with

common intracapsular conditions with pain will report that the pain occurs during function such

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as chewing and will have a positive response to loading the TMJ. In the current clinical case,

our patient did not respond to imposed loading of either TMJ and the duplication of pain (his

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chief complaint) could only be achieved when the patient opened maximally or protruded
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maximally causing condylar translation in both TMJs or by moving the mandible to the right side

causing translation of the condyle in the left TMJ. Deviation of pain complaints from the
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expected should be used as an indicator to more thoroughly evaluate potential sources of pain
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(see Table I).


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Another deviation from the norm observed in this clinical case was the high intensity of

pain during chewing. Most TMJ pain reports will be in the moderate range (VAS = 40-60) and

are usually lower than masticatory muscle pain (VAS = 50-70)10, 11. The patient was able to
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minimize his pain during times that he did not use the mandible and the intermittent pain, even
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though of high intensity, did not result in central sensitization12 (unmasking of inputs on second
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order neurons that result in a broad distribution of allodynia or pain from palpation). The very

limited ability to elicit pain in this case study supports the observation that his pain had not

progressed to promoting a central response even though he had multiple episodes of chewing

pain per day at a high intensity. The intermittent pain experience during chewing and the lack of

pain for significant intervals were important to minimize effects such as central sensitization.
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Central sensitization requires a persistent barrage of afferent input from peripheral structures

such as muscle or joint.13

The patient described his pain on the McGill Pain Questionnaire (MPQ) as sharp,

shooting and aching pain and this is a typical sensory description of pain from the TMJ with an

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anteriorly displaced disc or osteoarthritis. However, the MPQ affective and miscellaneous

descriptors chosen by the patient (Table I) are not commonly reported with temporomandibular

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joint pain14, particularly a pain that is intermittently experienced. These uncommon descriptors

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reported by this patient may have been secondary to the high intensity pain perception and

affective pain experience over a number of years.

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The sex of the patient may be another factor that may explain the pain experience of this

patient. In males, androgens such as testosterone have anti-nociceptive properties by


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stimulation of endogenous opioids15 and may have blunted the pain that was experienced with

the development of the bony lesion in the condyle. However, functional pain such as chewing
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elicited a high level of pain that may not be controlled by endogenous opiate release. In this
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clinical case, the age of the patient was early to middle 60’s when he started having pain and he
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most likely would have reduced circulating levels of testosterone that occurs with aging.

Although we do not know what the minimum level of testosterone that is required to affect pain
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perception, this patient may not have experienced as much reduction of perceived pain due to

androgens as compared to a younger male.


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CONCLUSIONS

It is important to recognize atypical pain presentations involving the temporomandibular

joint from more commonly encountered pains such as those associated with anteriorly displaced

discs (with or without reduction) or osteoarthritis. The clinician should evaluate each patient’s

pain presentation using standardized pain assessments such as Visual Analog Scales (VAS) or
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the McGill Pain Questionnaire16 to recognize these “red flags” as indicators of uncommon

temporomandibular joint pathophysiology.

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References

1. Boniello R, Gasparini G, D'Amato G, Di Petrillo A, Pelo S. TMJ metastasis: a unusual

case report. Head Face Med 2008;4:8.

2. Neville BWD, D. D.; Allen, C. M.; Bougout, J. E. Oral and Maxillofacial Pathology. 3rd

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Ed. ed. St. Louis, MO: Saunders/Elsevier; 2009.

3. Motamedi MH, Behroozian A, Azizi T, Nazhvani AD, Motahary P, Lotfi A. Assessment of

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120 maxillofacial aneurysmal bone cysts: a nationwide quest to understand this enigma.

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J Oral Maxillofac Surg 2014;72(8):1523-30.

4. Guarda-Nardini L, Piccotti F, Mogno G, Favero L, Manfredini D. Age-related differences

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in temporomandibular disorder diagnoses. Cranio 2012;30(2):103-9.

5. Lane NE, Brandt K, Hawker G, Peeva E, Schreyer E, Tsuji W, et al. OARSI-FDA


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initiative: defining the disease state of osteoarthritis. Osteoarthritis Cartilage

2011;19(5):478-82.
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6. Dijkgraaf LC, de Bont LG, Boering G, Liem RS. The structure, biochemistry, and
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metabolism of osteoarthritic cartilage: a review of the literature. J Oral Maxillofac Surg


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1995;53(10):1182-92.

7. Åkerman S, Rohlin M, Kopp S. Bilateral degenerative changes and deviation in form of


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temporomandibular joints An autopsy study of elderly individuals. Acta Odontologica

Scandinavica 1984;42(4):205-14.
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8. Magnusson C, Ernberg M, Magnusson T. A description of a contemporary human skull


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material in respect of age, gender, temporomandibular joint changes, and some dental

variables. Swedish Dental Journal 2008;32(2):69-82.

9. Wiese M, Svensson P, Bakke M, List T, Hintze H, Petersson A, et al. Association

between temporomandibular joint symptoms, signs, and clinical diagnosis using the

RDC/TMD and radiographic findings in temporomandibular joint tomograms. J Orofac

Pain 2008;22(3):239-51.
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10. McCreary CP, Clark GT, Merril RL, Flack V, Oakley ME. Psychological distress and

diagnostic subgroups of temporomandibular disorder patients. Pain 1991;44(1):29-34.

11. Plesh O, Curtis D, Levine J, McCall WD, Jr. Amitriptyline treatment of chronic pain in

patients with temporomandibular disorders. J Oral Rehabil 2000;27(10):834-41.

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12. Hu JW, Sessle BJ, Raboisson P, Dallel R, Woda A. Stimulation of craniofacial muscle

afferents induces prolonged facilitatory effects in trigeminal nociceptive brain-stem

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neurones. Pain 1992;48:53-60.

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13. Greene CS. Neuroplasticity and sensitization. J Am Dent Assoc 2009;140(6):676-8.

14. Mongini F, Italiano M. TMJ disorders and myogenic facial pain: a discriminative analysis

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using the McGill Pain Questionnaire. Pain 2001;91(3):323-30.

15. Fischer L, Arthuri MT, Torres-Chavez KE, Tambeli CH. Contribution of endogenous
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opioids to gonadal hormones-induced temporomandibular joint antinociception. Behav

Neurosci 2009;123(5):1129-40.
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16. Melzack R. The McGill pain questionnaire: Major properties and scoring methods. Pain
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1975;1:277-99.
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Figure Legends

Fig. 1. Panoramic film of patient with chewing pain and pain at maximum opening in the left

temporomandibular joint. A vague radiolucent area in the left mandibular condyle can be

observed (white arrow).

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Fig. 2. Corrected CBCT reconstructed images made perpendicular to the long axis of the

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condyle to visualize cross-sections of the left condyle from medial to lateral. The lesion (white

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arrow) can be easily identified in the series of images.

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Fig. 3. Coronal images of Technetium-99 scans demonstrate an active inflammatory process in

the left temporomandibular joint (white arrow). Image sequence represents scans progressing
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from superior to inferior through the head.
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Fig. 4. Representative microscopic image of the biopsy (H&E stain) recovered from the
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subchondral cyst of the left mandibular condyle. Dense fibrous tissue with mild chronic
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inflammation (white arrows) can be observed with non-viable entrapped cartilage (black arrow).
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Table I

Comparison of Typical and Atypical Symptoms and Signs of Temporomandibular Joint (TMJ)
Osteoarthritis

Subjective Typical Finding Atypical Finding (this case)

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History of TMJ sounds Crepitus Crepitus and clicking
History of pain during mandibular Chewing Pain only when opening
function wide such as yawning

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Pain description (MPQ*) Tiring Sharp
Troublesome Shooting
Nagging Blinding

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Sore Piercing
Tender Tight
Aching Dreadful
Pain intensity (VAS†) 40-60/100 90/100

Objective
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TMJ sounds (current) Crepitus Crepitus only at maximal
opening (45 mm)
Palpable tenderness lateral to TMJ Yes No
Pain with temporomandibular joint Yes No
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loading (biting on tongue blade on side


opposite to involved TMJ)
Vertical range of motion with pain Starts at 15-20 mm to Only at maximal opening of
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maximum opening (i.e., 45 mm (at the end of


during full range of condylar translation)
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condylar translation)
Horizontal range of motion with pain Full range of protrusion and Only at maximum
lateral excursion towards protrusion and maximal
opposite side of involved right lateral excursion
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temporomandibular joint
Muscle pain Yes No
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*McGill Pain Questionnaire


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Visual Analog Scale
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