Vol. 107 No. 6 June 2009

CLINICOPATHOLOGIC CONFERENCE

Editor: Paul C. Edwards

Painful deviation of the mandible Sergio Monteiro Lima Júnior, DDS,a Maximiana Cristina de Souza Maliska, DDS,b George Dimitroulis, DDS, MS,c Filipe Modolo, DDS, PhD,d and José Nazareno Gil, DDS, MS, PhD,e Florianópolis, Brazil; and Melbourne, Australia FEDERAL UNIVERSITY OF SANTA CATARINA AND UNIVERSITY OF MELBOURNE (Oral Surg Oral Med

Oral Pathol Oral Radiol Endod 2009;107:749-753)

CASE PRESENTATION Clinical presentation A 12-year-old girl was referred to the Oral and Maxillofacial Surgery Department by her orthodontist for evaluation of right facial asymmetry. Intraoral examination showed a mandibular dental midline deviated to the right with total disarticulation of the occlusion in the closed mouth position (Fig. 1). The patient reported difficulties during mastication, primarily pain involving the right temporomandibular joint (TMJ) and limited mouth opening. During opening movements, the mandible deviated to the right. The TMJs did not present sounds on auscultation. Her speech was normal. The patient did not report any history of facial trauma or previous TMJ treatment. Her lower first molars were lost as a result of caries. Her past medical history was noncontributory. Panoramic radiography did not reveal any alteration of the mandible. The condyles were of normal size and profile. An ovoid radiolucency adjacent to the right TMJ, in the area of the external auditory meatus, measuring approximately 5.0 ⫻ 5.0 mm was noted (Fig. 2). A similar image was not evident on the left side. Dental midlines were not coincident. Computerized tomography (CT) without contrast showed normal

a

Oral and Maxillofacial Surgeon, Department of Oral and Maxillofacial Surgery, Federal University of Santa Catarina. b Clinician, Department of Oral and Maxillofacial Surgery, Federal University of Santa Catarina. c Oral and Maxillofacial Surgeon, Department of Oral and Maxillofacial Surgery, University of Melbourne. d Oral and Maxillofacial Pathologist, Department of Pathology, Federal University of Santa Catarina. e Professor, Oral and Maxillofacial Surgery Department, and Chairman, Residence Program, Federal University of Santa Catarina. Received for publication Jul 4, 2008; returned for revision Dec 25, 2008; accepted for publication Jan 3, 2009. 1079-2104/$ - see front matter © 2009 Mosby, Inc. All rights reserved. doi:10.1016/j.tripleo.2009.01.002

condylar position at full closure, with lack of translational movements of the right condyle after mouth opening (Fig. 3). The soft tissue was not observed in this exam. The CT did not reveal any other alterations.

Differential diagnosis The important clinical features that stand out in this case are the patient‘s age, the apparent facial asymmetry, the occlusal disarticulation, the history of pain, and the deviation to the right during opening movements. The differential diagnosis of facial asymmetries and limited mouth opening should include unilateral disc displacement without reduction or the anchored disc phenomenon (both causing a unilateral closed lock), unilateral mandibular dislocation, condylar hyperplasia, coronoid hyperplasia, unilateral TMJ fibrous ankylosis, condyle osteoarthritis, and primary or metastatic tumors of the condyle. The panoramic radiograph and CT images excluded condylar hyperplasia, coronoid hyperplasia, unilateral TMJ fibrous ankylosis, condyle osteoarthritis, and tumors of the condyle in this particular case. Closed lock may be caused either by an internal derangement of the TMJ or by the anchored disc phenomenon. Closed lock is clinically recognized as an inability of the patient to open the mouth. Disc displacement without reduction is caused by laxity of the lateral disc attachment that allows migration of the disc to an anterior and medial position, resulting in a mechanical barrier to the movement of the condyle. Disc displacement without reduction presents with a restriction of translation movement, absence of clicking, deviation toward the affected side on opening the mouth, limitation in lateral movement toward the contralateral side, and restriction in protrusive movements, with the mandible shifting toward the affected side. Pain is present on palpation and during opening movements. The anchored disc phenomenon is characterized by a sudden-onset and persistent closed lock of the TMJ. This phenomenon occurs when the disc is pressed against the fossa in the absence of sufficient lubrication.1 Opening of

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Fig. 2. Panoramic radiograph did not reveal any alteration of the mandible. The condyles were of normal size and profile. The radiolucent area near the right condyle (arrow) was thought to represent the external auditory meatus.

Fig. 1. A, Frontal view of the patient showing right facial asymmetry. B, Intraoral view, showing general disarticulation of the occlusion and deviation of the mandibular midline to the right in the closed position.

the mouth is associated with deviation of the mandibular midline toward the affected side. Contralateral movements are limited, and ipsilateral movements are normal. Protrusive movements are restricted, with the mandible shifting toward the affected side. The patient complains of pain if mandibular opening is forced. The initial limitation in mouth opening is severe and inflexible, and it improves only slightly over prolonged periods. Temporomandibular joint dislocation is a condition in which the condyles slide beyond the articular eminence, resulting in locking. This condition affects between 3% and 7% of the population,2 and it can be classified as acute, chronic, or recurrent. Clinically, the patient presents with an open lock, functional impairment, facial deformity, and pain. The etiology of mandibular dislocation includes intrinsic or extrinsic trauma resulting in rupture, tearing, or stretching of the TMJ ligaments and capsule, hyperfunction of the protractor muscles, and degeneration of the TMJ ligaments and capsule. Unilateral dislocation causes facial asymmetry and mandibular deviation, producing discomfort and pain during mastication and speech.

Fig. 3. Three-dimensional computerized tomography reconstruction. A, Condyles in normal position at full closure. An occlusal disarticulation is seen. B, On full mouth opening, the right condyle rotates (arrow) and the left condyle performs a complete translation (arrowhead).

The ovoid radiolucency noted on the panoramic radiograph most likely represented superimposition of the ear cartilage or the external auditory meatus. Nevertheless, because of the

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Fig. 4. A, Sagittal view (T2-proton density–weighted magnetic resonance imaging [MRI]) shows a hyperintense image suggestive of a cystic lesion behind the right condyle. B, T1-Proton density–weighted MRI demonstrates an ovalshaped lesion posterior to the right condyle (arrow). Note the condyle in front of the lesion and the auditory meatus behind it.

patient’s complaint, the possibility that it represented a pathologic lesion, such as synovial chondromatosis, a temporomandibular joint cyst, a lesion originating from the parotid gland, or a benign neural mass, was also considered. Synovial chondromatosis of the temporomandibular joint is a rare, benign, tumor-like disorder. It is characterized by chondrometaplasia of the synovial membrane. Cartilaginous nodules are formed and may become pedunculated and detached from the synovial membrane, forming loose bodies within the joint space.3 Many patients present with temporomandibular dysfunction, usually monoarticular, that may be misinterpreted as a tumor of the parotid gland. Ganglion cysts and synovial cysts of the TMJ are rare. These lesions are similar in that they may be asymptomatic and patients are often only aware of the presence of a swelling in the preauricular region. There may be pain and an obvious cosmetic deformity; some patients also complain of trismus.4 Imaging exams should differentiate these from lesions of the parotid gland. The 2 most common tumors of the parotid gland are the pleomorphic adenoma and papillary cystadenoma lymphomatosum (Warthin tumor). Pleomorphic adenoma typically appears as a painless, slow-growing, and firm mass. It has female predilection and is usually found in the fifth decade of life. Papillary cystadenoma lymphomatosum usually presents as a single nodular, well defined, slow-growing, firm to soft mass in the parotid region. It is painless, movable, and completely encapsulated. It is diagnosed at a mean age of 62 years.5 Palisaded encapsulated neuromas, schwannomas, and neurofibromas are benign tumors originating from superficial nerves. These lesions are common in the head and neck regions and usually present as painless nodules, although tenderness or pain may be noted in some instances.

Fig. 5. Scintigraphy revealed an area of contrast uptake, suggestive of a cyst, near the right condyle (arrow).

Fig. 6. Occlusion after muscle relaxion.

Diagnosis and management Magnetic resonance imaging (MRI) was performed to study the relation between the disc and condyle of the temporomandibular joints, revealing a 1.5 ⫻ 1.5 mm oval and well circumscribed hyperintense image posterior and lateral to the right condyle (Fig. 4). The signal was uniform throughout the mass and compatible with a cystic lesion. There was no apparent relationship between the lesion and the parotid gland. Scintigraphy was performed to rule out condylar hyperplasia, corroborating the radiographic diagnosis of a cyst (Fig. 5).

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Fig. 7. Histopathology (hematoxylin and eosin). A, Myxoid degeneration of connective tissue of the capsule of the TMJ (original magnification ⫻100). B, Presence of flattened fibroblasts lining the cavity (original magnification ⫻400).

The patient was admitted to the hospital for surgical removal of the lesion. During general anesthesia, a postural mandibular asymmetry was diagnosed, because the surgeon was manually able to return the mandible to the midline position in the absence of trismus (Fig. 6). A preauricular incision was made to approach the TMJ and remove the lesion. The parotid gland was elevated because the lesion was beneath its capsule, near the posterior pole of the condyle. There was no communication of the cyst with the joint. During dissection, the mass ruptured, allowing the escape of a gelatinous material. Postoperative recovery was uneventful. The mandibular shift to the right side was no longer evident and the patient was no longer in pain. Follow-up examination 4 months later showed no recurrence of the mandibular deviation. The excised tissue was fixed in 10% neutral buffered formalin and embedded in paraffin. Histologic examination showed a cyst-like structure lined by fibrous connective tissue without synovial lining cells and containing eosinophilic amorphous material (Fig. 7). The final diagnosis was ganglion of the TMJ.

DISCUSSION Ganglions of the TMJ are mucin-filled pseudocysts which are thought to develop as a result of myxoid degeneration of the connective tissue of the capsule of the joint.6 The true synovial cyst of the TMJ develops as a result of herniation of the synovium into the surrounding tissues or displacement of the synovial tissue during embryogenesis.7 On histologic examination, a ganglion does not contain any epithelial lining, whereas a synovial cyst presents with synovial or endothelial cells lining the cyst cavity.4 The clinical presentations of these lesions are similar. Only histologic examination can differentiate between them. Since the first report in 1977,8 no case series of ganglions or synovial cysts of the TMJ have been

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reported. We reviewed the 27 cases of ganglia and synovial cysts of the TMJ published in the Englishlanguage literature.4,6-29 It is interesting to note that no reports of recurrence were found in the literature, probably because this lesion is developmental in origin and contains a thick fibrous wall that can be completely dissected from surrounding tissues. Of the cases reviewed, 17 were ganglions and 9 were synovial cysts. One case report did not include a histologic description.17 The median age of patients was 42 years, with a higher incidence of synovial cyst after the fourth decade and of ganglions after the fifth decade. Men presented an equal distribution between synovial cyst and ganglion (3:3), whereas women exhibited a prevalence of ganglion over synovial cyst (13:6). Two articles did not report gender.9,17 The synovial cyst was seen more commonly in the right TMJ (6:2), whereas the ganglion was almost equally distributed between right and left sides (9:10). Except for 1 case,23 all reported facial enlargement or asymmetry. The mean size of the lesions was 20 mm. Pain was the most common presenting symptom, reported in 20 cases.4,6-8,10-13,15,16,18-24,29 Other signs and symptoms included condylar resorption,14,15,24 crepitation,15,26 clicking,16,29 and calcifications.20 Three reports described bone involvement, including an intraosseous ganglion,11 an intraosseous synovial cyst associated with otorrhea,25 and an extension of the synovial cyst through the temporal bone.23 Kim et al.27 analyzed the contents of a ganglion of the TMJ, identifying proteins originating from bacteria as well as dystroglycan, a glycoprotein that forms part of a protein complex which links the extracellular matrix to the cytoskeleton. The presence of dystroglycan might be due to degenerative changes of the TMJ.27 Panoramic and plain films typically fail to demonstrate TMJ cysts and ganglions,7,19,20 although they are still indicated to evaluate for osseous pathology. In the present case, although examination of the panoramic radiograph revealed an apparent radiolucency near the right condyle, this image likely represented the external auditory meatus. The fact that a ganglion cyst was ultimately discovered in this area appears to have been coincidental. Computerized tomography (CT) is recommended because it reveals the relationship between the lesion and the mandibular condyle.15 Singer17 suggested using MRI to confirm the clinical suspicion of TMJ cyst and to exclude a parotid gland lesion. Kerawala28 stressed the importance of clinical examination for surgical planning because the MRI may not reveal the full extent of these lesions. Ultrasound can also be helpful in ruling out parotid gland involvement.6 In the past, treatment often involved superficial parotidectomy,9-11 because these lesions were mistakenly

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thought to be associated with the parotid gland. With MRI, it is now possible to precisely locate and to preoperatively determine the cystic nature of these lesions. Treatment involves complete surgical removal, preserving the neighboring structures, such as the facial nerve and the parotid gland.24,26-28 No recurrences are expected. The present case presented with facial asymmetry caused by a postural shift of the mandible to the right. The CT imaging did not reveal a definite mandibular asymmetry, and the mandible could be returned to the midline position during general anesthesia. The occlusal disarticulation noted in Figs. 1 and 3 represented the most comfortable position the patient could achieve. In the panoramic radiograph, it is possible to notice a dental midline discrepancy with the condyles apparently in a normal position. It is interesting to note this patient was able to shift her mandible to the right without moving the left condyle from the articular fossa. The appearance of the lesion on MRI established a cyst of the TMJ as the most likely diagnosis. The ganglion reported here probably did not present clinically as a preauricular mass because of its small size and its deeper location in relation to the temporal fascia and posteriorly to the condyle. REFERENCES 1. Nitzan DW, Etsion I. Adhesive force: the underlying cause of the disk anchorage to the fossa and/or eminence in the temporomandibular joint—a new concept. Int J Oral Maxillofac Surg 2002;31:94-9. 2. Kendall BD, Booth PW. Surgical correction of temporomandibular dislocation. Atlas Oral Maxillofac Surg Clin North Am 1996;04:278-86. 3. Balliu E, Medina V, Vilanova JC, Pelaez I, Puig J, Trull JM, et al. Synovial chondromatosis of the temporomandibular joint: CT and MRI findings. Dentomaxillofac Radiol 2007;36:55-8. 4. Silva EC, Guimaraes AL, Gomes CC, Gomez RS. Ganglion cyst of the temporomandibular joint. Brit J Oral Maxillofac Surg 2005;43:77-80. 5. Eveson JW, Cawson RA. Warthin’s tumor (cystadenolymphoma) of salivary glands: a clinicopathologic investigation of 278 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1986;61:256-62. 6. Lopes V, Jones JA, Sloan P, McWilliam L. Temporomandibular ganglion or synovial cyst? A case report and literature review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1994;77:627-30. 7. Chang YM, Chan CP, Kung Wu SF, Hao SP, Chang LC. Ganglion cyst and synovial cyst of the temporomandibular joint: two case reports. Int J Oral Maxillofac Surg 1997;26:179-81. 8. Heydt S. A ganglion associated with the temporomandibular joint. J Oral Surg 1977;35:400-1. 9. Janecka IP, Conley JJ. Synovial cyst of temporo-mandibular

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