British Journal of Oral and Maxillofacial Surgery (2000) 38, 205208 2000 The British Association of Oral and

d Maxillofacial Surgeons doi:10.1054/bjom.1999.0181

BRITISH JOURNAL OF ORAL

& M A X I L L O FA C I A L S U R G E RY

Synovial chondromatosis of the temporomandibular joint

H. Miyamoto, * H. Sakashita, D. F. Wilson, A. N. Goss *Former Visiting Research Fellow, Oral & Maxillofacial Surgery Unit; Currently Department of OMFS & Dentistry, Head, Department of OMFS & Dentistry, Fujieda Municipal General Hospital, Fujieda, Shizuoka, Japan; Associate Professor, Oral Pathology; Professor & Director, Oral & Maxillofacial Surgery Unit, The University of Adelaide, Adelaide, Australia SUMMARY. We report two cases of temporomandibular joint (TMJ) synovial chondromatosis, one of which was in the early stage and treated arthroscopically. The second was more advanced and recurred after removal of the free bodies, and so required condylectomy and synovectomy.

INTRODUCTION Synovial chondromatosis commonly affects the knee, elbow, wrist, shoulder and hip; involvement of the temporomandibular joint (TMJ) is rare.2,3 The main symptoms are pain, swelling, and clicking with deviation and limitation of mandibular movement.4 Because TMJ synovial chondromatosis is so rare, it is often mistaken for an internal derangement which has similar symptoms.3 Computed tomography (CT) and magnetic resonance imaging (MRI) can show loose bodies in the joint space, and are excellent investigations if synovial chondromatosis is suspected.5,6 Double contrast arthrotomography will help to diagnose nodules within the joint space,7 and direct inspection of the joint space by arthroscopy will confirm the diagnosis.8 The histological metaplastic changes of synovial chondromatosis can be separated into three phases of development:9 phase 1 (early) intrasynovial involvement with metaplasia only in the synovial membrane

and no loose bodies; phase 2 (transitional) intrasynovial involvement and free bodies with metaplasia in the synovial membrane, and detached particles; and phase 3 (advanced) multiple free bodies varying in size from 110 mm or more together with detached particles and no sign of intrasynovial disease. In the earliest phase, there is proliferation of undifferentiated stem cells in the stratum synoviale.9 Later, cells that resemble fibroblasts form a primitive chondroid matrix from which the resulting nodules of cartilage grow, either by proliferation of the interstitial cell that produced the chondroid matrix or by surface growth of new cells that differentiate near the surface to produce the matrix.9 In this paper, we report the diagnosis and management of two cases of TMJ synovial chondromatosis, one early and one transitional. CASE REPORTS CASE 1
A 21-year-old woman presented to the Dentistry and Oral Surgery Clinic at the Ishikawa Prefectural Central Hospital complaining of pain in the right TMJ for the past 15 months. On physical examination, her maximal mouth opening was 34 mm, which caused pain that was constant at 80 on visual analogue scale (VAS, 0100) in the right TMJ. When she opened her mouth her mandible deviated to the right with crepitus. She also had a history of squamous cell breast cancer (T3N2MO) which had been diagnosed several months before she presented to us. She had responded well to the operation but had a poor prognosis. Panoramic and transcranial radiographs and coronal tomograms showed bony changes on the condyle of the right side but no calcifying lesions in the right TMJ. MRI (T1 weighted images) showed two masses of low signal intensity anteriorly in the right TMJ with bony changes in the condyle (Fig. 1). These findings suggested a clinical diagnosis of TMJ osteoarthrosis and anterior disc displacement without reduction of the TMJ. There was no sign of metastatic disease in her mandible. Conservative treatment (physiotherapy and
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Fig. 1 Preoperative MRI (T1) of the right TMJ (case 1) showing two masses of low signal intensity (big arrows) anterior to the bony changes in the condyle (small arrow).

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Fig. 2 Operative arthroscopic view of the anterior upper space of the TMJ (case 1) showing metaplasia (arrows) only in the synovial membrane (A and B).

Fig. 3 Histological appearance of the synovial metaplasia (case 1). (A) Low-power photomicrograph showing hyperplastic, folded synovial lining (L). The hypercellar synovial tissues merge with less cellular fibrous tissue and pseudo cartilage (C). (S=joint space; haematoxylin and eosin; original magnification 40); (B) High-power photomicrograph showing hyaline cartilage-like tissue. (haematoxylin and eosin; original magnification 400).

anti-inflammatory drugs) had been tried for three months previously, but she had not responded. We did a double puncture arthroscopic examination of the right TMJ under general anaesthesia, which showed metaplasia in the synovial membrane of the upper joint space (Fig. 2) and a section of the synovium around the metaplasia looked fibrillated. There were pseudowall capsular adhesions with synovitis in the anterior compartment, but no loose bodies. Under arthroscopic guidance, the metaplasia and grossly abnormal synovium were removed and the capsular adhesions resected. The patient was immediately able to open her mouth to 50 mm with slight deviation to the right side and her pain was recorded as 0 (VAS). The postoperative MRI showed that the mass of low signal intensity had been removed (Fig. 3). On histological examination, the specimens showed focal, chronic proliferative synovitis with early metaplasia of the

cartilage (Fig. 4). These findings confirmed the diagnosis of early synovial chondromatosis of the TMJ. The patient has been followed up regularly for the past 15 months and has been symptom-free with no recurrence.

CASE 2
A 37-year-old woman presented to a private specialist in Adelaide with a 10-year history of intermittent clicking, locking, and painful right TMJ that had severely limited her jaw movements for the last six months. She had initially been successfully treated with a bite splint by a prosthodontist eight years previously. No radiographs had been taken at that time. On physical examination, she had a maximum mouth opening of 15 mm with pain and crepitations in the right TMJ. She was otherwise fit and well, and there was no

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Fig. 4 Postoperative MRI (T1) of the right TMJ (case 1) showing that the mass has been removed.

Fig. 6 Histological appearance of the synovial metaplasia (case 2). (A) Low-power photomicrograph showing two partially calcified hyaline cartilage bodies (C). These cartilage bodies lie adjacent to or are confluent with dense fibrous connective tissue (F) (haematoxylin and eosin; original magnification 400); (B) High-power photomicrograph of a cartilage body exhibiting patchy calcification (arrows). The calcifying cartilage is partially circumscribed by a layer of non-vital focally calcified cartilage (NV). (haematoxylin and eosin); (original magnification 200).

Fig. 5 Preoperative CT (case 2) showing calcifications in the right TMJ (arrows).

history of injury to the jaw. A CT showed some calcifying masses in the right TMJ (Fig. 5), consistent with synovial chondromatosis. At arthrotomy under general anaesthesia, there were multiple calcifications in the upper joint space of the right TMJ, and 20 milky-white loose bodies were removed. Because the joint space and the synovium were clear and looked normal to the naked eye, they were preserved. As her mouth opening was only 25 mm at the time, we did a bilateral coronoidectomy to produce a final mouth opening of 35 mm. Subsequently, her mouth opening reduced to 23 mm by four months and to 18 mm by six months. A CT was taken eight months postoperatively which showed recurrence of the synovial chondromatosis and fibrous ankylosis of the right TMJ, so we did a condylectomy and synovectomy

under general anaesthesia nine months after the initial operation, which increased her mouth opening to 45 mm. On histological examination, the loose bodies that had been removed at the first operation were composed of multiple nodules of hyaline cartilage covered by fibrous connective tissue. The tissue that was removed at the second operation showed cartilaginous metaplasia (Fig. 6). These findings confirmed second phase transitional synovial chondromatosis of the TMJ. The patient could open her mouth to 40 mm four months postoperatively and has been followed up for 10 months with no recurrence.

DISCUSSION Of our two cases of synovial chondromatosis of the TMJ, one was phase 1 of the disease and the second in phase 2 (transitional). In case 1 there was pronounced metaplasia in the synovial membrane on the MRI of the displaced and grossly altered disc. Arthroscopic examination confirmed the diagnosis. In case 2 CT confirmed the diagnosis in that there were loose bodies in the TMJ. Various treatments have been used for synovial chondromatosis of the TMJ.2,8 When arthrography

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British Journal of Oral and Maxillofacial Surgery 4. Forssell K, Happonen R-P, Forsell H. Synovial chondromatosis of the temporomandibular joint: report of a case and review of the literature. Int J Oral Maxillofac Surg 1988; 17: 237241. 5. Ingen JM van, Man K de, Bakri I. CT diagnosis of synovial chondromatosis of the temporomandibular joint. Br J Oral Maxillofac Surg 1990; 28: 164167. 6. Igarashi C, Kobayashi K, Imanaka M et al. Image findings of synovial chondromatosis of temporomandibular joint: report of eight cases and review of literature. J Jpn Stomatol Soc 1996; 45: 462469. 7. Miyamoto H, Sakashita H, Miyata M, Kurita K. Arthroscopic diagnosis and treatment of temporomandibular joint synovial chondromatosis: report of a case. J Oral Maxillofac Surg 1996; 54: 629631. 8. Carls FR, Hochstetter A von, Engelker W, Sailer HF. Loose bodies in the temporomandibular joint: the advantages of arthroscopy. J Craniomaxillofac Surg 1995; 23: 215221. 9. Milgam JW. Synovial chondromatosis: a histopathologic study of thirty cases. J Bone Joint Surg 1977; 59A: 792801. 10. Miller AS, Hardwock RD, Daley DJ. Temporomandibular joint synovial chondromatosis: report of a case. J Oral Surg 1978; 36: 467468. 11. Coolican MR, Dandy DJ. Arthroscopic management of synovial chondromatosis of the knee: findings and results in 18 cases. J Bone Joint Surg 1989; 71B: 498500. 12. Swan EF, Owens WF. Synovial chondromatosis: a case report with spontaneous regression and review of the literature. South Med J 1972; 65: 14961500.

confirms its location in the upper space, it can be removed by arthroscopic surgery,7 otherwise open surgery or arthrotomy for removal of the loose bodies with the addition of partial or complete synovectomy is required.8,10,11 In the first or second phase, removal of the mass together with all loose bodies and the affected synovium is usually sufficient8 because the recurrence rate is low and it may even regress spontaneously.13 If direct examination at the time of surgery shows only loose bodies with inactive synovial disease (the third phase) synovectomy is not necessary.9 Case 1 was treated arthroscopically because the condition was early and confined to the superior joint space, and the patients general prognosis was poor. The lesion was removed arthroscopically and this was confirmed by postoperative MRI. In case 2 the synovial chondromatosis recurred eight months after the first operation. The first operation was aimed at removing the loose bodies and sparing the joint structure as it was thought that the disease was advanced. The synovium looked normal to the naked eye, but histologically the synovial membrane was metaplastic. This patients maximum incisal opening decreased from 23 mm at four months to 18 mm at six months postoperatively. The development of fibrous ankylosis on the lateral pole of the joint is consistent with some surgical articular damage as well as recurrence of the synovial chondromatosis. In patients with synovial chondromatosis whose mouth-opening decreases postoperatively recurrence should be considered. Further surgery should be aggressive and include synovectomy for the chondromatosis. Condylectomy may be required to overcome the ankylosis.
References
1. Fee WE Jr, Windorst P, Wiggins R, Pang L. Synovial chondromatosis of the temporomandibular joint. Otolaryngol Head Neck Surg 1979; 87: 741748. 2. Wise DP, Ruskin JD. Arthroscopic diagnosis and treatment of temporomandibular joint synovial chondromatosis: report of a case. J Oral Maxillofac Surg 1994; 52: 9093. 3. Mendonca-Caridad JJ, Schwartz HC. Synovial chondromatosis of the temporomandibular joint: arthroscopic diagnosis and treatment of a case. J Oral Maxillofac Surg 1994; 52: 624625.

The Authors
H. Miyamoto Former Visiting Research Fellow Oral & Maxillofacial Surgery Unit; Currently Department of OMFS & Dentistry H. Sakashita Head, Department of OMFS & Dentistry, Fujeida Municipal General Hospital, Japan D. F. Wilson Associate Professor Oral Pathology University of Adelaide, Adelaide, Australia A. N. Goss Professor & Director Oral & Maxillofacial Surgery Unit University of Adelaide, Adelaide, Australia Correspondence and requests for offprints to: Professor A.N. Goss, Oral & Maxillofacial Surgery Unit, University of Adelaide, South Australia, 5005 Paper received 26 October 1998 Accepted 1 February 1999