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SYNOVIAL CHONDROMATOSIS AFFECTING THE TEMPOROMANDIBULAR JOINT CASE REPORT A N D LITERATURE REVIEW PHILLIP S. ROSEN, K E N N E T H P. H. P R I T Z K E R , JOSEPH G R E E N B A U M , R I C H A R D C . HOLGATE, and ARNOLD M. NOYEK A case of synovial chondromatosis affecting the temporomandibular joint is reported and the literature is reviewed. Chondromatosis occurs most frequently in this joint in middle-aged women, and it presents with pain and tenderness over the joint. Radiologically, the condition is detected by widening of the joint space with the variable presence of radiodense loose bodies. Pathologically, the loose bodies in the case presented were demonstrated to arise from cartilaginous metaplasia of the synovial lining. The stimulus for synovial chondrometaplasia at this site is unknown. Synovial chondromatosis is a rare condition in which chondroid bodies develop within the synovium and a r e extruded into the joint space. These cartilaginous structures are commonly thought to arise from From the Departments of Medicine. Pathology, Radiology, Otolaryngology. and the Rheumatic Disease Unit. Mount Sinai Hospital and University of Toronto. Toronto, Ontario. Canada. Phillip S . Rosen, M.D., F.R.C.P. (C): Staff Physician. Mount Sinai Hospital. Associate Professor, University of Toronto: Kenneth P. H. Pritrker, M.D.. F.R.C.P. (C): Pathologist. Mount Sinai Hospital. Assistant Professor, University of Toronto: Joseph Greenbauni. M.D.. F.R.C.P. (C): Fellow in Rheumatology, Mount Sinai Hospital and University of Toronto: Richard C. Holgate. M.D., F.R.C.P. (C): Staff Neuroradiologist. Mount Sinai Hospital. Lecturer. University of Toronto; Arnold M. Noyek. M.D., F.R.C.S. (C): Staff Otolaryngologist, Mount Sinai Hospital. Assistant Professor. University of Toronto. Address reprint requests to Kenneth P. H. Pritzker. M.D.. Department of Pathology. Mount Sinai Hospital. 600 University Avenue. Toronto. Ontario. Canada M5G 1x5. Submitted for publication July 20, 1976: accepted September 17. 1976. Arthritis and Rheumatism, Vol. 20, No. 2 (March 1977)

metaplasia of the synovium rather than from neoplastic processes (1-3). Although this condition is usually associated with large joints such as the knee, hip, or shoulder (4,5). it may also affect the temporomandibular joint (6-15). T h e purpose of this case report is t o draw attention to this entity in the differential diagnosis of pain and swelling of the temporomandibular joint.

CASE REPORT M J . a 44-year old housewife, presented with a 4-month history of left ear pain and swelling of the left pre-auricular area. There was no history of trauma or joint locking. Physical examination revealed a poorly defined, fixed, tender swelling, 2.5 crn in diameter, anterior to the left tragus (Figure I ) . There was no pain on movement of the jaw or restriction of jaw motion. Radiologic examination revealed normal bone scan a n d skull films. Multidirectional tomography of the left temporomandibular joint showed widening of the joint space, sclerotic erosive changes on the cranial side of the joint, and anterior spur formation on the mandibular condyle. Several loose bodies were detected within the joint space (Figure 2 ) . Because of the persistent mass. the joint was explored at operation. On opening the joint, multiple, small, whitish masses were removed from the joint cavity. Histologic examination showed that the loose bodies consisted of hyaline cartilage (Figure 3 ) with some cytologic atypia of the chondrocytes (Figure 4). Fine deposits of calcium were demonstrated by Von Kossa stain throughout the cartilaginous bodies (Figure 4). A few fragments of synovium were also identified, and chondroid metaplasia of the synovial cells was observed (Figure 5 ) . Postoperatively, the patient experienced jaw pain aggravated by movement. Because of persistence of this pain, 4

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months later she was readmitted t o the hospital and underwent excision of the left mandibular condyle. Histologic examination of the condyle showed that the normal fibrocartilaginous articular surface was absent and that the articular surface consisted of sclerotic bone. N o hyaline cartilage was observed within the condyle. Postoperatively, the patient was asymptomatic.

DISCUSSION A review of the literature (Table 1 ) indicates that the present patient is typical of the 12 cases previously reported in clinical presentation and operative findings (6-15). Eleven of the 13 cases were female, most cases were in patients over 40, and most cases present with pain in the temporomandibular joint. Only 6 of 13 cases presented with a definite mass. That the lesion was radiologically defined in only 5 of the 13 cases may reflect the fact that in most of the cases the loose bodies consisted of cartilage only, and therefore were relatively radiolucent. In the cases in which loose bodies were seen on the x ray, calcification of the cartilage or bone formation was demonstrated in the structures histologically. In this particular patient, detection of the slightly calcified loose bodies radiologically depended on the high resolution afforded by multidirectional tomography.

Fig 1. A clinical photograph of the patient demonstrates a diSJuse .swelling approsiiiiatelv 2.5 X 2.5 em overlying the left temporomandibular .joirit.

Fig 2. A. A n anterior-posterior multidirectional tomogram demonstrates sphenoid fossa sclerosis and multiple radiodeti.vitie.s (arrows) within the It$ temporoniandibular joint. B. A lateral multidirectional tomugram receals a radiodense mass (arrow) within the retnporomandibular ,joint space, accompanied bj, marked .sclerosis of the glenoid ,/6.ssa.

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Fig 4. Plio/oi,ric,rogrup/i o,/ a reprr.sentatice loose hod!. s h o ~ : rpleornorphisni o f the chondror,'te.v. Henlaro.y.viin and ro.viri / X 631. The inset depicts fine!,, partirulale calrificatiori in the cartilage inatris. ).'on Ko.s.su .slclirl / x 3 ~ 0 , .

SYNOVIAL CHONDROMATOSIS

739

Fig 5. f'hotoniicrograph of the .yynoviurn demonstrates increased density of the jibrillar stronia about the synorial lining cells. tieriiator,.lin and eosin ( X 63 1. The inset .shows a s.pnovial cell undergoing chondroq ~ i rire/apla.~ia c (arrow). Hernatoxylin and eosin ( X 300).

With the exception of the cases reported by Trevor (7), Kusen (lo), and the second case of Schneider (9). the material usually removed from the temporomandibular joint consisted of relatively few loose bodies. This fact raises the question of whether the intraarticular loose bodies may in fact be a part of another

disease, such as osteochondritis dissecans or osteoarthritis, or secondary to previous fracture. Most patients had no history of trauma. In osteochondritis dissecans or osteoarthritis, it would be expected that the loose bodies would be representative of the tissue normally present on the articular

Table 1. Svnovial Chondromatosis of the Ternporornandibular Joint Case and Reference

Age

Sex

Pain

Swelling

+

-

Axhausen (6) 1933 Trevor (7) 1952 Feist ( 8 ) 1960 Schneider (9) Case I 1960

21 63 59 52

F F F F

+ +

5. Schneider (9) Case 2 1960 6. Kusen (10) 1969 7. Schulte(l1) 1969 8. Silver(l2) 1971 9. Ballard ( I 3) Case I I972 10. Ballard ( I 3) Case 2 1972 1 1 . Alling (14) 1973 12. Tasanen(l5) 1974 13. Present case 1976

47 28 31 18 42 37 53 40 44

F F F M M F F F F

+ + + + + + + +

I. 2. 3. 4.

+ +

-

Loose Bodies Number of on X Rays Loose Bodies

-

++

-

+ + +

-

Numerous 48 I Numerous (not removed) 80 480 I 5 1

I 3 3 15

ROSEN ET AL

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surface. It is an anatomic peculiarity of the temporomandibular joint that hyaline cartilage is absent from the articular surface in the adult (16,17). T h e formation of hyaline cartilage nodules in the temporomandibular joint cavity therefore indicates either a metaplastic or neoplastic process. T h e presence of chondroid metaplasia within the synovium in our cases, a n d the absence of any further chondroid material with resection of t h e mandibular condyle are evidence that at least in the present case the loose bodies arose from synovial metaplasia. Again, the osteoarthritic changes in the mandibular condyle may be a result of the chondromatosis rather than an etiologic factdr, because over 50% of reported cases with synovial chondromatosis in other joints have associated secondary osteoarthritic changes (4). N o recurrent cases of chondromatosis involving the temporomandibular joint have been reported, possibly because of the complete removal of all the loose bodies at the time of operation, t h e small amount of synovium in the temporomandibular joint, o r the relatively short follow-up times in the individual case reports. Temporomandibular joint chondromatosis, like chondromatosis in other joints, predominantly affects patients from 30 to 60 years of age (3,5,18). However, although 65-69% of patients with chondromatosis involving more commonly affected joints such as the knee or hip are male ($1 8), 75% of t h e patients with temporomandibular joint chondromatosis reported in this review are female-a striking difference in sex incidence. Nonetheless, chondromatosis of the temporomandibular joint closely resembles chondromatosis in other joints in predominant symptoms, pain, a n d swelling (5,18), in the variable presence of multiple radio-opaque joint masses (5,l8), a n d in the microscopic appearance of cartilaginous o r osteocartilaginous joint loose bodies with synovial chondrometaplasia ( 1-3,5). Synovial chondromatosis is an infrequent condition, but one that must be considered in the differential diagnosis of pain a n d swelling of the temporomandibular joint. Radiographically, widening of the joint spaces and presence of loose bodies are the diagnostic criteria of this condition. However it should be emphasized that the absence of loose bodies on radiologic examination does not rule out this diagnosis. Excision of the loose bodies and-if symptoms from secondary osteoarthritic changes persist-removal of the mandibular condyle appear to b e effective treatment for this disease.

ACKNOWLEDGMENTS The authors thank Mr. I. Gareau and the Division of Instructional Media, Mount Sinai Hospital, for preparing the photographs, and Ms. Beth Sinclair for typing and retyping the manuscript.

REFER EN CES 1. Jones H T Loose body formation in synovial osteochondromatosis with special reference to the etiology and pathology. J Bone Joint Surg 6:407-458, 1924 2. Freud E: Chondromatosis of the joints. Arch Surg 34~670-686, I937 3. Spujut HJ, Dorfman HD, Fechner RE, et al: Lesions of

4. 5.

6. 7.

8. 9. 10.

Synovial origin, tumors of bone and cartilage, Atlas of Tumor Pathology. Second series. Fascicle 5, 1970, pp 391-410 Mussey RD, Henderson MS: Osteochondrosis. J Bone Joint Surg 31:619-627, 1949 Trias A, Quintana 0: Synovial chondrometaplasia: review of world literature and a study of 18 Canadian cases. Can J Surg 19:151-158, 1976 Axhausen G: Pathologie und Terapie des Kiefergelenkes. Fortschr Zanheilk 9: 184, 1933 Trevor D: A case of synovial chondromatosis of the temporomandibular joint. Postgrad Med J 28: 408-409, 1952 Feist JH, Gibbons TG: Osteochondromatosis of the temporomandibular joint. Radiology 74:291-293, 1960 Schneider G : Chondromatosis des Kiefergelenkes. Dtsch Zahnaerzt Z 3:1233, 1960 Kusen GJ: Chondromatosis: report of case. J Oral Surg

27:735-738. 1969 I I . Schulte WC, Rhyne RR: Synovial chondromatosis of the temporomandibular joint. Oral Surg 28:906-913, 1969 12. Silver CM, Simon SD, Lichman HM, et al: Synovial chondromatosis of the temporomandibular joint. J Oral Surg 31~604-606, 691-693, 1973. 13. Ballard R , Weiland LH: Synovial chondromatosis of the temporomandibular joint. Cancer 30791-795, 1972 14. Alling CC, Rawson DW. Staats OJ. et al: Synovial chondromatosis of the temporomandibular joint. J Oral Surg 31:604-606. 691-693, 1973 15. Tasanen A, Lamberg MA, Kotilainen R: Osteochondromatosis of the temporomandibular joint. Oral Surg 38:845-849. 1974 16. Wright DM, Moffett BC: The postnatal development of

the human temporomandibular joint. Am J Anat I4 1:235-249, 1974 17. Moffett BC, Johnson LC, McCabe JB, et al: Articular remodeling in the adult human temporomandibular joint. Am J Anat 115:119-142, 1974 18. Murphy FP, Dahlin D, Sullivan CR: Articular synovial chondromatosis J Bone Joing Surg 44A:77-86, 1962