35fe1d01-sinovial kondromatosis tmj

8/3/2019 35FE1d01-Sinovial Kondromatosis TMJ

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CASE REPORT

Synovial Chondromatosis of the TemporomandibularJoint: An Asymptomatic Case Report and Literatu reReviewDenis Pimenta e Souza. D.D.S.; Caio Cesar de Souza L oureiro. 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.

0886-9634/2801-067S05.00/0, THEJOURNAL OFCRANIOMANDIBULARPRACTICE,Copyr ig t i t2010by CHROMA, Inc.

Manuscript receivedAugust 19, 2008; acceptedJanuarv 7, 2009Address for correspondence:Dr. Ricardo RaitzAv. Heitor Peneteado, 1832,101/ACEP: 05438-300Sumarezinho. Sao Paulo-SPB r a z i lE-mail: [email protected]

ABSTRACT: Synovial chondromatosis of the temporomandibular joint (TMJ) is a rare lesion character-ized 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 com mon characteristics,although this combination is not always apparent. Radiopacities of the TMJ should be thoroughly inves-tigated as some signals and symptoms may be not present or combined, taking months or even yearsto confirm a diagnosis. A case report is presented here w ith a brief literature review, whe re surgicalremoval was the therapy of choice, calling attention to the absence of symptoms and some signals,which may mislead final diagnosis.

Dr. Denis Pimenta e Souza is a postgraduate student in the oral and rtuLxillo-fitciat program. School of Dentistry.University of Sao Paulo, an d an oral andmaxillofaciai surgery assistant professorin the Section of Oral am i Maxillofacia!Surger\; Hospital Santa Paula, SaoPaulo, Brazil.

Synovial chondromatosis is an uncommon benignmon oailicular arthropathy characterized by the for-mation of multiple cartilaginous or osteocartilagi-nous metaplastic nodules in synovial and subsynovialcon nec tive tissu e nf the Jo int s.' '" It most frequen tlyaffects the large an icular joints such as knee, hip, elbow,shoulder, and wrist.""'-^ Although, the involvement ofthe temporomandihular joint (TMJ) is rare, many caseshave been published since 1933. when Georg Axhausenreported ihe first case.'-'"'- '^Osteocartil agi nous loose b odies of TMJ can arise as adirect result of the proliferative disorder of the sinovium(sinovial chondromatosis). or secondary to osteochondraifractures or osteoarthritis'*'- ' ' ' ' '-! ' (secondary sinovialchondrometaplasia). The primary form seems to be moreaggressive and bone erosive and probably originates frommesenchymal remnants that become mataplastic. calcify,and break off into the joint space. The secondary form isassociated with degenerative, inflammatory and nonin-flammatory diseases and is a more passive process.-"*Swelling, unilateral pain, occlusal changes, clicking,crepitation, deviation, and limited mandibular functionare the most common characteristics, although this com-bination is not always apparent.i'''i"i''i''i Since the syn-


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SYNOVIAL CHONDROMATOSIS OF THE TMJ SOUZA ET A

ovial chondromatosis of the TMJ is a rare condition.these features may be easily misdiagnosed as neoplasia orother pathologies/'-1*Imaging diagnoses includes conventional x-ray exami-

nation, computed tomography (CT). and magnetic reso-nance imaging {MRI). Recently, arlhro.scopy has heenused as a more conservative means of ohlaining a defini-tive diagnosis''"' and removing loose bodies when iheyare small enough for the instrument.'' '-'' Arthrotomy orthe surgical removal of the loose bodies, with or withoutresection of the synovial membrane and disk--^'-'^ arestill largely used therapies, as they dispense with usingexpensive equipment and allow seeing and biopsyingcritically the pathologic tissues. Here is presented a casereport where the surgical removal was the therapy ofchoice, calling attention to the absence of symptomswhich may mislead fmal diagnosis.Case Report

A 28-year-old man was referred to the Section of Oraland Maxillofacial Surgery at the Hospital Santa Paula(Sao Paulo. Brazil) by his orthodontist who first noticedsome radiopaque particles in the region of ihe right TMJ,through an orthodontic documentation.On clinical examination, no evidence of facial asym-metry or malocclusion was noticed. There was no limita-

tion of mandibular movement nor mandibular deviationduring mouth opening (Figure 1). Swelling and crepita-tion were noticed while palpating the right TMJ. Thepatient denied any history of trauma to the maxillofacialregion, lntraorally. it was noted the absence of several

Figure INo evidence of fcial asym-iiielry, no limitalion ofmandibukir movement duringmouth opening iiitiveiTieni.nor mandibular deviatitm.

teeth and severe peHodontitis.Conventional panoramic radiography demonstrated 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 scarevealed the presence of multiple round-shaped, highdensily masses, with aspect of loose bodies, located neathe right temporal eminence occupying the joint spacwhere the disk should be positioned (Figure 3).

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

These clinical and image findings led us to a diagnotic hypothesis of synovial chondromatosis. It was decideto access the glenoid fossa surgically in order to take biopsy of the affected tissues or only remove the loosbodies.

After induction of general anesthesia, the TMJ aninfratemporal fossa were approached via modified preau

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


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SOUZA ET AL. SYNOVIAL CHONDROMATOSIS OF THE TMJ

ricular incision (Figure 4). White, irregularly shapedloose bodies escaped from the TMJ upper space after thejoint capsule was opened (Figure 5). The glenoid fossawas explored, and the idherent cartilaginous mass freedfrom its attachments to the fossa walls. All the loosebodies were removed (Figure 6), Since the condyle andthe disk were macroscopically normal, condilectomy andmenisceclomy were not indicated and so closure wasobtained with preservation ofthe synovium, capsule, andcondyle.

Figure 4 preauricuiurlo approachintratemporal cavity.

Figure 6IiTcgularly shaped, w hile, canilaginous nodules removed from joinlcomparlmenl al surgery.

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

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

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


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SNYOVIAL CHONDROMATOSIS OF THE TMJ SOUZA ET AL

DiscussionThis case of Synovial chondromatosis is considereduncommon us large articular joints such as knee, hip.

elbow., shoulder, and wrist usually are mostly affectedinstead of the TMJ.i-'"^-'-"-i-' For the characteristicsassessed, one could conclude that this is a primary formof the pathology, which is represented as a benign carti-laginous metaplasia of mesenchymal tissue with rem-nants arising in the synovial membrane where fibroblastsbeneath its surface becom e metaplastic and deposit chon-dromucin. Thus, a cartilaginous focus is stimulated, andonce formed, it grows by active cellular proliferation.'**Once the cartilaginous metaplastic and calcified nod-u|gs4.7.14.15 ^[^;e from the synovial membrane, as well asfrom the fibrocartilaginous disk tissue, they extrude to thejoint sp ace as loose bod ies, often surrounded by fibrosedconnective tissue where they are nourished by the syn-ovial fluid.'-'' '"^"^ occ upying the joint space where thedisk should be positioned, and usually causing pain.Surprisingly, this patient was asymptomatic and Ihclesion was rst noticed through an orthodontic documen-tation. Moreover, only preauricular sw elling, and crepita-tion were present in this case. These few characteristicsmay lead this lesion to be misdiagnosed as neoplasia,especially chondrosarcoma'- or other pathologies such asdegenerative joint disease, rheumatoid arthritis , neu-rotrophic arthritis, tuberculosis, and osteochondritis ossi-f icans . ' ' ' Therefore , imaging examinat ions such asradiographies. CT scanning, and MRI must be carried outfor a correct diagnosis and therapy.' '-"'''Radiographie appearance is variable and may includewidening of the joint space, manifestations of degenera-tive changes t)f the articular surfaces, and expansion ofthe joint capsule, but evidence of loose bodies is notalways present, being found in only 60^ ofthe cases.""'This was the case herein reported, where radiopaque par-ticles could be seen into the glenoid fossa (Figure 2). CTplays an important ro!e in the diagnosis of the TMJ syn-

ovial chondromatosis, since it can demonstrate soft tissueswelling, possible change of the articular surface of thetemporal bone, and define size, shape, and locations ofthe loose calcified bodies-"'-'*' (Figure 3). However. MRIis mostly used to establish the expansion and thickeningof the joint capsule and morph ologic changes in the posi-tion ofthe disk."^Immunohistopathologicai studies have shown that dif-ferent growth factors and hormones may play an impor-tant role in the patJiogenesis of synovial chondromatosis.Fujita, et al.^ reported that Transforming Growth Factor (TGF-) and Tenascin (TN) were strongly present in thesynovial 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 iimportant for chondrogenesis in the extracellular matriand the condensing mesenchyme of developing boneThese findings support the metaplastic theory of synoviachondrom atosis in the TMJ since n either TG F nor TN arnormally present in the synovial membrane of normajo in t s . Sato , et al. ' " reported that different fibroblasgrowth factors (FGF) and their respective receptor(FGFR) may be strongly related to the deveiopment osynovial chondromatosis. In their study. FGF-2 anFGFR-1 immunoreactivities were observed in chondrocytes while FGFR-.^ and its specific iigand, FGF-9, werimmunohistochemically observed at the margins of thcartilage nodules. It was concluded that expression oFGFR-1 in chondrocytes contributes to the growth potent ia l of synovia l chondromatos is , and tha t the FGF2/FGFR -l system may play an important role, as well athe FGF-9/FGFR-3 system, in its pathogcnesis.

Recently, arthroscopy of the TMJ. by providing tissufor a histomorphologic analysis, has been used as a morconservative means of obtaining the definitive diagnosand definitive treatment."'^ " However, the technique difficult to execute; patients still have to suffer the surgcal damage resultant from the insertion ofth e arthroscopinto the joint c avity , ' and some loose bo dies are bienough to inable this technique. M oreover, not all the sugical services dispose from the equipment. Various othetreatments have been used, For a long time, completremoval ofthe synovium associated or not with condylectomy or condylotomy was the main therapy for thpathology.- Nowadays, this radical approach is rarelindicated. More conservative procedures such as arthrotomy and removal oft he loose bodies, partial or total synovectomy, and. particularly, if both joint compartmenare affected or if the disk is damaged beyond functionarepair, diskectomy are the treatment of choice.''-""'In the case reported, the surgical approach enabled th

removal of either little or big loose bodies. As no signifcant alterations were found in the synovium. disk ocondyie. a very conservative surgical procedure wataken, with no need of synovium or disk removaRadiopacities of the TMJ should be thoroughly investgated as some signals and symptoms may be not presenor combined, taking months or even years to confirm diagnosis.'"References

1. Miindriiili S. Pulilo J. IJtm-s SA. Cliiuscr L; Synovial choinlromalnsisof ilemphm.yc>i"wi.(i-,Vn/-,i, ' 2W1: 18:1486-1488,2. B e l le Sharp CW. Kourie LR. Hmchinson D; Conservavc surgical nianag


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SOUZA ET AL. SNYOVIAL CHONDROMATOSIS OF THE TMJ

meni of synovial chonilromatosis. Oral Surg Oral Med Oral Pathol OralRadio! Endnd. 1997: 4:592-593.Fujita S. li/uka T,Yoshida H. Segami N: Transforming growth factor andtenascin m synovial chondromatosis of the teriiporomndibular joint,Repon()faca.ie./'i. /Ora/AuV/(f/i;ci'ws 1997; 26:258-2^9.Ikebe T. Nakayama E,Shinohara M, Takeuchi H. Takenoshita Y: Synovialeliondromatosis ofthe [emporomandihular joint: the effectof interlcukin-lon loose-body-derived cells. Oral Surg Oral Med Oral Pathol Oral RadialWtnn998: 85:526-531.Karli V. Giickman RS. Zavlow M: Synovia! chondromatosis of the leni-pommandibularjoinf with intracranial extension. Ora/5ur ; , 'Or/MP /Ora /PatUiil Oral Radial Endod 1998: 86:664-666.M endonea-Cari dad JJ, Schwan/. HC: Synovial chondromatosis of ihe tem-porimiandibular joint: arthroscopic diagnosis and treatment of a case. JOral MiLxUhfac Siirn 1994: 52:624-625.Petito AR, Bennett J, As. ael LA. Carlotti AE Jr.: Synovial chondromatosis ofthe temporomandibular joint: varying presentation in four ca.ses. Orfl/iSur^Oral Med Oral Pnthol Oral Radio! Endod 2{XK): 90:758-764.P-iimopoulou M. Karakasis D. Magnudi D,Tirou V. Eleftheriadis I:.Synovial chondromatosi!. of the temporomandibular joint, lir J OralMtLxillofac SuvK 1998: .16:317-.I8.Reinish El, Feinberg SE. Devaney K: Primary synovial chotidromatosis ofthe tenipommandibularjoinl with su.'jpected triuimatic etiology. Report of acase. Int .1 Oral Maxillofac Siirg 1997: 26:419-422.Saio J, Segami N. Suzuki T, Voshitake Y. Nishikawa K: The expression offibn>bla.st growtii factor-2 atid fiiinibla.'it growth (actor receptor-1 in chon-drocytcs in synovial c h un drum a los i s of the temporomandihular joint.Report oftwo eases. Int J Oral Miuillofac Surf; 2002: 31.532-536,Holmlunt!AB . Eriksson L. Reinboli FP: Synovia! chondromatosis of the tem-p>romandihutar joint: clinical, surgical and histological aspects. Int J OralMa.\!lhfac SurR 2003: 32:143-147.Miyamoto H. Saka.shita H, Miyata M. Kuriia K: Arthroscopic diagnosis andtrciitment oftemporomandibular joint synovia! ehiindromattwis: rcpiirt ofacase. J Oral MiLxillofac Sur,; 1996: 54:629-631,von Lindem JJ, Theuerkauf I. Niederhagen B. Berge S. Appel T.Reich RH:Synovial chondromatosis of the temporumandibular joini: clinical, diag-nostic, and hist OUI oqihologic findings. Oral Surg Oral Med Oral PatholOral Radini Endod 20U2; 94:31 -38,

14, Felices RR. Giordano OA: Cotidromatosis sinovial de la ATM, A.soc15, Wise DP. Ruskin JD: ,\rthri)scopic diagnosis and treatment of temporo-mandibular joint synovial chondromatiisi.s: report of a case, ,/ Oral

Mivitllofai Suri; 1994: 52:9()-9.',16, Lucas JH. Quinn P, Foote J, Baker S. Brtino J: Recurreni symivial chondro-malosis treaieii with meniscectomy and synovectomy. Onit Sur^ OralMed Oral Pathol Oral Radiol Endod 997:84:253-258,17, Mgram JW: Thecla,ssincal!on ofloose bodies in human joints. Clin Orthop1977: 124:282-291.

I a. .Xiang S, Rebellato J. Inwards CY. Keller EE: Malocclusion as.sociated withosteocartil agi nous loo.sc bodies ofthe temporomandihular joitit. J Am DentAssoc 21X15: 136:484-489.19, Yu 0 . Yang J, Wayng P. Shi H. Luo J: CT features of synoviui chondro-matosis ill the icmptirotnandibularjoinl. Oral Surg Oral Med Oral PutlwlOral Radio! Endod 2004; 97:524-528,

Dr. Caio Cesa r de Souza Loureiro is an oral and maxilhfaeial resi-dent in the Section of Oral and Mcilhfacial Surgery, Hospital SantaPaula. 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 SantaPatda. .Si'io Paulo. Brazil.

Dr. Luis Fernando lAtho Leandro is chief of the Section of'Oral andMiLxittofacial Surt;ery. Hospital Santa faida. Sao Paulo, Brazil.

Dr. Ricardo Raitz is a professor o f the Biodentistry post graduateiM.Sc.iprogriim of Ihirapuera University. Sao Paulo. Brazil: professorand chair of General Pathology al Sdo Caetano do Sul Univers ily. SaoPaulo, Brazil.


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