Atypical temporomandibular joint pain: a case report

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. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

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Authors:

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Manuscript Title: Atypical Temporomandibular Joint Pain: A Case Report

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, College of Medicine, University of Florida

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Address correspondence to:

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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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Charles G. Widmer, D.D.S., M.S. Department of Orthodontics Box 100444, JHMHSC College of Dentistry University of Florida Gainesville, FL 32610-0444 Phone: 352-273-5696 Fax: 352-846-0459 e-mail: [email protected]

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,

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

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temporomandibular joint is indicated. The following case report provides details of one patient with an unusually high level of TMJ pain only at maximum opening and atypical pain descriptors

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associated with his pain.

CASE REPORT

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

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pain and a history of pain in his left temporomandibular joint (TMJ) area only while chewing and opening wide during yawning. This pain started approximately five years prior to being 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 pain was rated at an intensity of 90/100 on a Visual Analog Scale (VAS) and was only elicited when he was opening his jaw wide during yawning. A positive history of reciprocal clicking in

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

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

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

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

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

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they are irregular with a “moth-eaten” appearance2. Additionally, the prolonged duration of this patient’s lesion is inconsistent with the rapid growth typically seen in malignant 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 connective tissue lining. Pain and swelling are common; however, crepitus is rare. Aneurysmal bone cysts typically affect patients under the age of 303, which places this

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condition lower on the differential diagnosis for this patient. The age of the patient and the presence of distinct crepitus within the TMJ would

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suggest that the most commonly encountered diagnosis of osteoarthritis may be a contributing factor for his pain4. The reciprocal clicking was consistent with an anteriorly 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

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hard or soft tissue examination of the patient.

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

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bony remodeling and to explore the potential for an active infection, further diagnostic testing was pursued using skeletal scintigraphy with correlative gallium imaging. An increase in 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 of the scintigraphy study were consistent with a reactive process. The patient was subsequently

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

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

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were removed and submitted for pathological examination and interpretation. The results of the biopsy were consistent with dense fibrocollagenous tissue with fragments of non-viable 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

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damage that results from stresses that may be initiated by an abnormality in any of the synovial joint tissues, including articular cartilage, subchondral bone, ligaments, menisci (when present), peri-articular muscles, peripheral nerves or synovium”5. TMJ osteoarthritis (OA) has been described as a four-stage pathogenesis: (1) initial and repair; (2) early; (3) intermediate; and (4)

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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 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 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 degenerative changes in the TMJ and, based on logistic regression, found that age and sex (females) were predictors of degenerative changes with age as the most dominant predictor9.

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

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the development of TMJ osteoarthritis whereas older age has a much higher relationship. The presentation in this reported case is consistent with late stage OA. Pain was elicited 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

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include disc displacement with or without reduction, arthridities and post-traumatic injuries. 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 without the need for advanced diagnostic techniques such as imaging. Most of the patients with

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common intracapsular conditions with pain will report that the pain occurs during function such as chewing and will have a positive response to loading the TMJ. In the current clinical case,

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our patient did not respond to imposed loading of either TMJ and the duplication of pain (his chief complaint) could only be achieved when the patient opened maximally or protruded 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 (see Table I).

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

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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 though of high intensity, did not result in central sensitization12 (unmasking of inputs on second 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,

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shooting and aching pain and this is a typical sensory description of pain from the TMJ with an 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 reported by this patient may have been secondary to the high intensity pain perception and

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affective pain experience over a number of years.

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 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 clinical case, the age of the patient was early to middle 60’s when he started having pain and he most likely would have reduced circulating levels of testosterone that occurs with aging.

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

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androgens as compared to a younger male.

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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temporomandibular joint pathophysiology.

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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

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observed (white arrow).

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

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

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

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 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 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

Objective TMJ sounds (current)

Crepitus Yes Yes

Starts at 15-20 mm to maximum opening (i.e., during full range of condylar translation) Full range of protrusion and lateral excursion towards opposite side of involved temporomandibular joint Yes

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Palpable tenderness lateral to TMJ Pain with temporomandibular joint loading (biting on tongue blade on side opposite to involved TMJ) Vertical range of motion with pain

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Pain intensity (VAS†)

Tiring Troublesome Nagging Sore Tender Aching 40-60/100

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Muscle pain

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Horizontal range of motion with pain

*McGill Pain Questionnaire †

Visual Analog Scale

Atypical Finding (this case) Crepitus and clicking Pain only when opening wide such as yawning Sharp Shooting Blinding Piercing Tight Dreadful 90/100

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Typical Finding Crepitus Chewing

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Subjective History of TMJ sounds History of pain during mandibular function Pain description (MPQ*)

Crepitus only at maximal opening (45 mm) No No

Only at maximal opening of 45 mm (at the end of condylar translation) Only at maximum protrusion and maximal right lateral excursion No

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