CASE REPORT

Synovial Chondromatosis of the Temporomandibular Joint: An Asymptomatic Case Report and Literature Review
Denis Pimenta e Souza. D.D.S.; Caio Cesar de Souza Loureiro. D.D.S.; Paula Felix Falchet. D.D.S.; Luiz Fernando Lobo Leandro. D.D.S.. Ph.D.; Ricardo Raitz, D.D.S., Ph.D.

0886-9634/2801067S05.00/0, THE JOURNAL OF CRANIOMANDIBULAR PRACTICE, Copyrigtit2010 by CHROMA, Inc.

Manuscript received August 19, 2008; accepted Januarv 7, 2009 Address for correspondence: Dr. Ricardo Raitz Av. Heitor Peneteado, 1832, 101/A

ABSTRACT: Synovial chondromatosis of the temporomandibular joint (TMJ) is a rare lesion characterized by the presence of loose bodies in the glenoid fossa. Swelling, unilateral pain, occlusal changes, clicking, crepitation, deviation, and limited mandibular function are the most common characteristics, although this combination is not always apparent. Radiopacities of the TMJ should be thoroughly investigated as some signals and symptoms may be not present or combined, taking months or even years to confirm a diagnosis. A case report is presented here with a brief literature review, where surgical removal was the therapy of choice, calling attention to the absence of symptoms and some signals, which may mislead final diagnosis.

CEP: 05438-300
Sumarezinho. Sao Paulo-SP

Brazil
E-mail: ricardorartz@ig.com.br

S
Dr. Denis Pimenta e Souza is a post graduate student in the oral and rtuLxillofitciat program. School of Dentistry. University of Sao Paulo, and an oral and maxillofaciai surgery assistant professor in the Section of Oral ami Maxillofacia! Surger\; Hospital Santa Paula, Sao Paulo, Brazil.

ynovial chondromatosis is an uncommon benign monoailicular arthropathy characterized by the formation of multiple cartilaginous or osteocartilaginous metaplastic nodules in synovial and subsynovial connective tissue nf the Joints.' '" It most frequently affects the large anicular joints such as knee, hip, elbow, shoulder, and wrist.""'-^ Although, the involvement of the temporomandihular joint (TMJ) is rare, many cases have been published since 1933. when Georg Axhausen reported ihe first case.'-'"'- '^ Osteocartil agi nous loose bodies of TMJ can arise as a direct result of the proliferative disorder of the sinovium (sinovial chondromatosis). or secondary to osteochondrai fractures or osteoarthritis'*'-'''''-!' (secondary sinovial chondrometaplasia). The primary form seems to be more aggressive and bone erosive and probably originates from mesenchymal remnants that become mataplastic. calcify, and break off into the joint space. The secondary form is associated with degenerative, inflammatory and noninflammatory diseases and is a more passive process.-"* Swelling, unilateral pain, occlusal changes, clicking, crepitation, deviation, and limited mandibular function are the most common characteristics, although this combination is not always apparent.i'''i"i''i''i Since the syn-

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ovial chondromatosis of the TMJ is a rare condition. these features may be easily misdiagnosed as neoplasia or other pathologies/'-1* Imaging diagnoses includes conventional x-ray examination, computed tomography (CT). and magnetic resonance imaging {MRI). Recently, arlhro.scopy has heen used as a more conservative means of ohlaining a definitive diagnosis''"' and removing loose bodies when ihey are small enough for the instrument.'' '-'' Arthrotomy or the surgical removal of the loose bodies, with or without resection of the synovial membrane and disk--^'-'^ are still largely used therapies, as they dispense with using expensive equipment and allow seeing and biopsying critically the pathologic tissues. Here is presented a case report where the surgical removal was the therapy of choice, calling attention to the absence of symptoms which may mislead fmal diagnosis. Case Report A 28-year-old man was referred to the Section of Oral and Maxillofacial Surgery at the Hospital Santa Paula (Sao Paulo. Brazil) by his orthodontist who first noticed some radiopaque particles in the region of ihe right TMJ, through an orthodontic documentation. On clinical examination, no evidence of facial asymmetry or malocclusion was noticed. There was no limitation of mandibular movement nor mandibular deviation during mouth opening (Figure 1). Swelling and crepitation were noticed while palpating the right TMJ. The patient denied any history of trauma to the maxillofacial region, lntraorally. it was noted the absence of several

teeth and severe peHodontitis. Conventional panoramic radiography demonstrated a radiopaque mass into the glenoid fossa of the right temporal bone and around the head of the right condyle. which showed no deformity (Figure 2). A CT scan revealed the presence of multiple round-shaped, highdensily masses, with aspect of loose bodies, located near the right temporal eminence occupying the joint space where the disk should be positioned (Figure 3).

Figure 2 Pre-operativc piinoraniic radiography revealed radiopacities in the area ot the right TMJ.

These clinical and image findings led us to a diagnostic hypothesis of synovial chondromatosis. It was decided to access the glenoid fossa surgically in order to take a biopsy of the affected tissues or only remove the loose bodies. After induction of general anesthesia, the TMJ and infratemporal fossa were approached via modified preau-

Figure I No evidence of fcial asymiiielry, no limitalion of mandibukir movement during mouth opening iiitiveiTieni. nor mandibular deviatitm.

Figure 3 Axial computed tomography scan demonstrating multiple high-density masses around ihc right TMJ.

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ricular incision (Figure 4). White, irregularly shaped loose bodies escaped from the TMJ upper space after the joint capsule was opened (Figure 5). The glenoid fossa was explored, and the idherent cartilaginous mass freed from its attachments to the fossa walls. All the loose bodies were removed (Figure 6), Since the condyle and the disk were macroscopically normal, condilectomy and menisceclomy were not indicated and so closure was obtained with preservation ofthe synovium, capsule, and condyle.

Figure 4 preauricuiur lo approach intratemporal cavity.

Figure 6 IiTcgularly shaped, while, canilaginous nodules removed from joinl comparlmenl al surgery.

Postoperatively, the patienl displayed decreased pain and swelling, but some little limitation on mandibular range of motion was noticed for 15 days. At a two-year follow-up, the mandibular range of motion continues to he normal, and the patient has had no symptoms. Radiographic examination showed no signs of recurrence (Figure 7).

Figure 5 L*K)se bodies migrated from the upper compartment after incision of ihc capsule.

Figurv 7 Posl-operativc panurmic radingraphv al iwo-years follow-up, showing no recurrence nf the lesion.

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Discussion This case of Synovial chondromatosis is considered uncommon us large articular joints such as knee, hip. elbow., shoulder, and wrist usually are mostly affected instead of the TMJ.i-'"^-'-"-i-' For the characteristics assessed, one could conclude that this is a primary form of the pathology, which is represented as a benign cartilaginous metaplasia of mesenchymal tissue with remnants arising in the synovial membrane where fibroblasts beneath its surface become metaplastic and deposit chondromucin. Thus, a cartilaginous focus is stimulated, and once formed, it grows by active cellular proliferation.'** Once the cartilaginous metaplastic and calcified nodu|gs4.7.14.15 ^[^;e from the synovial membrane, as well as from the fibrocartilaginous disk tissue, they extrude to the joint space as loose bodies, often surrounded by fibrosed connective tissue where they are nourished by the synovial fluid.'-'' '"^"^ occupying the joint space where the disk should be positioned, and usually causing pain. Surprisingly, this patient was asymptomatic and Ihc lesion was rst noticed through an orthodontic documentation. Moreover, only preauricular swelling, and crepitation were present in this case. These few characteristics may lead this lesion to be misdiagnosed as neoplasia, especially chondrosarcoma'- or other pathologies such as degenerative joint disease, rheumatoid arthritis, neurotrophic arthritis, tuberculosis, and osteochondritis ossificans.''' Therefore, imaging examinations such as radiographies. CT scanning, and MRI must be carried out for a correct diagnosis and therapy.' '-"''' Radiographie appearance is variable and may include widening of the joint space, manifestations of degenerative changes t)f the articular surfaces, and expansion of the joint capsule, but evidence of loose bodies is not always present, being found in only 6 0 ^ ofthe cases.""' This was the case herein reported, where radiopaque particles could be seen into the glenoid fossa (Figure 2). CT plays an important ro!e in the diagnosis of the TMJ synovial chondromatosis, since it can demonstrate soft tissue swelling, possible change of the articular surface of the temporal bone, and define size, shape, and locations of the loose calcified bodies-"'-'*' (Figure 3). However. MRI is mostly used to establish the expansion and thickening of the joint capsule and morphologic changes in the position ofthe disk."^ Immunohistopathologicai studies have shown that different growth factors and hormones may play an important role in the patJiogenesis of synovial chondromatosis. Fujita, et al.^ reported that Transforming Growth Factor (TGF-) and Tenascin (TN) were strongly present in the synovial membrane and in the extracellular matrix of the

synovial intima, respectively. TGF seems to increase differentiation of mesenchymal cells, production of proteoglycans. and replication of chondroblasts. while TN is important for chondrogenesis in the extracellular matrix and the condensing mesenchyme of developing bones. These findings support the metaplastic theory of synovial chondromatosis in the TMJ since neither TGF nor TN are normally present in the synovial membrane of normal joints. Sato, et al.'" reported that different fibroblast growth factors (FGF) and their respective receptors (FGFR) may be strongly related to the deveiopment of synovial chondromatosis. In their study. FGF-2 and FGFR-1 immunoreactivities were observed in chondrocytes while FGFR-.^ and its specific iigand, FGF-9, were immunohistochemically observed at the margins of the cartilage nodules. It was concluded that expression of FGFR-1 in chondrocytes contributes to the growth potential of synovial chondromatosis, and that the FGF2/FGFR-l system may play an important role, as well as the FGF-9/FGFR-3 system, in its pathogcnesis. Recently, arthroscopy of the TMJ. by providing tissue for a histomorphologic analysis, has been used as a more conservative means of obtaining the definitive diagnosis and definitive treatment."'^ " However, the technique is difficult to execute; patients still have to suffer the surgical damage resultant from the insertion ofthe arthroscope into the joint cavity,' and some loose bodies are big enough to inable this technique. Moreover, not all the surgical services dispose from the equipment. Various other treatments have been used, For a long time, complete removal ofthe synovium associated or not with condylectomy or condylotomy was the main therapy for this pathology.- Nowadays, this radical approach is rarely indicated. More conservative procedures such as arthrotomy and removal ofthe loose bodies, partial or total synovectomy, and. particularly, if both joint compartments are affected or if the disk is damaged beyond functional repair, diskectomy are the treatment of choice.''-""' In the case reported, the surgical approach enabled the removal of either little or big loose bodies. As no significant alterations were found in the synovium. disk or condyie. a very conservative surgical procedure was taken, with no need of synovium or disk removal. Radiopacities of the TMJ should be thoroughly investigated as some signals and symptoms may be not present or combined, taking months or even years to confirm a diagnosis.'" References
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Dr. Caio Cesar de Souza Loureiro is an oral and maxilhfaeial resident in the Section of Oral and Mcilhfacial Surgery, Hospital Santa Paula. Sao Paulo. Brazil. Dr. Paula Felix Falchct ii an oral and tnuxdtofacial .surgery as.si.^an! professor, St'clion of Oral and Maxillofacial Surgery, Ho.'ipiial Santa Patda. .Si'io Paulo. Brazil. Dr. Luis Fernando lAtho Leandro is chief of the Section of'Oral and MiLxittofacial Surt;ery. Hospital Santa faida. Sao Paulo, Brazil. Dr. Ricardo Raitz is a professor of the Biodentistry post graduate iM.Sc.iprogriim of Ihirapuera University. Sao Paulo. Brazil: professor and chair of General Pathology al Sdo Caetano do Sul Universily. Sao Paulo, Brazil.

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