Departamento financiero

Stefano Mandrioli, MD, Jessica Polito, MD,

Stefano Andrea Denes, MD, Luigi Clauser, MD, DMD, PhD

Ferrara, Italy

Abstract:Synovial chondromatosis is a cartilaginous metaplasia of the mesenchymal remnants of the synovial tissue of the joints. Its main characteristic is the formation of cartilaginous nodules in the synovium and inside the articular space (loose bodies). Synovial chondromatosis mainly affects big synovial joints such as the elbow and knee and is uncommon in the temporomandibular joint. The main symptoms are pain, limitation of jaw movement, crepitation, and inflammation. Diagnosis is made by panoramic radiograph, computed tomography scan, and mainly magnetic resonance imaging. Surgery is the therapeutic choice. The authors describe their experience in the treatment and in the follow up of a patient with unilateral synovial chondromatosis.

Key Words: Synovial chondromatosis, temporoman- dibular joint, cartilaginous metaplasty

S

ynovial chondromatosis (SC) is a rare condition in which there is cartilaginous metaplasia of the mesenchymal remnants of the synovial tissue of the joints, which is more of an active metaplastic than a neoplastic process. Synovial chondromatosis was first described by Ambroise ParO` in 1558 and is a cartilaginous metaplasty of mesenchymal remnants of synovial tissue of the joints. It is characterized by the formation of cartilaginous nodules in the syno- vium and inside the articular space (loose bodies).The first description of SC in the temporomandibular joint (TMJ) was published by Axhausen in 1933.1

It usually affects single large synovial joints such as the knee, hip, or elbow, but the TMJ may also be affected. Approximately less than 80 cases are described in the literature2,3with one case of bilateral involvement described. TMJ SC has a frequency four times higher in females4with a medium age of 55 and rarely occurs in younger people. The most common clinical signs and symptoms are unilateral pain, inflammation, swelling over the joint, limitations of the movement of the jaw, and crepitation in the joint. The diagnosis is achieved by panoramic radiograph,

computed tomography, magnetic resonance ima- ging, TMJ arthroscopy, and arthrography.2

CLINICALREPORT

A 50-year-old woman was referred to the Depart- ment of Cranio-Maxillo-Facial Surgery of St. Anna Hospital and University. She reported pain and clicking of the right TMJ for 3 months with moderate swelling of the preauricular area. The mouth opening was 35 mm with right lateral deviation and pain was evoked during palpation of the TMJ.

Diagnostic imaging was performed with pano- ramic radiograph, computed tomography, and mag- netic resonance imaging (Fig 1).

Computed tomography scan and magnetic resonance imaging showed the presence of multiple nodules inside the articular cavity of the right TMJ with no dislocation of the disc. Distension of the lateral capsule and fluid in the joint were very sug- gestive of SC. The patient was submitted to surgery under general anesthesia.

Preauricular incision (Fig 2) was carried out with exposure of the articular capsule. The distended lateral capsule was incised and both upper and lower compartments were explored. A large number of loose bodies were carefully removed from the upper compartment (Fig 3); the disc in the right position with no alterations was left in place. Synovectomy in areas with marked inflammation (red and hyper- plastic) was performed.

Histopathologic examination showed an inflam- matory infiltrate containing several multinucleated

From the Unit of Cranio-Maxillo-Facial Surgery, Centre for Craniofacial Deformities and Orbital Surgery, Ferrara, Italy.

Address correspondence and reprint requests to Dr. Luigi Clauser, Unit of Cranio-Maxillo-Facial Surgery, Centre for Cra- niofacial Deformities and Orbital Surgery, Corso Giovecca, 203, 44100 Ferrara, Italy; E-mail: [email protected]

Fig 1 Preoperative magnetic resonance image of the right temporomandibular joint.

giant cells and a focal osteoid formation was found, but there were no mitoses or cellular atypia indicat- ing malignancy.

The patient started articular physiotherapy 2 days later. A dental splint was then applied to improve TMJ function and masticatory muscles relaxation. After 1 year, the patient reported pain in the right preauricular area with limited mouth opening. A magnetic resonance image was per- formed and showed no recurrence (Fig 4).

Arthrocentesis was then performed and articu- lar lavage allowed the resolution of pain and limited mouth opening. A Michigan splint was then applied for a period of 6 months with resolution of the symptoms.

DISCUSSION

Three stages of the SC evolution have been de- scribed.5The first includes metaplasia of the synovial membrane with proliferation of nondifferentiated

cells without free bodies. The second stage is a progressive metaplasia that has a slow onset of loose body formation, which take off from the synovium. These bodies contain active chondrocytes. The third and advanced phase leads to degeneration with calcification of the loose bodies. Without knowing the cause, SC involves only the superior compart- ment of the joint and the few cases in which loose bodies were found inside the inferior compartment were the result of a perforation of the articular disc and the subsequent migration of these bodies from the superior compartment.2Y6

Phytogenesis is still unknown. Traumatisms, parafunction, and infections have been suggested but they do not seem to be the main cause.2,6Y8_Some other studies suggest the involvement of the fibro- blast growing factor and fibroblast growing factor receptor.9

Diagnosis of SC is based on clinical radiographic and histologic findings.8,9

The differential diagnosis must be made with osteochondritises, condylar tumors, avascular necro- sis, arthritis, or condylar fractures.

A recent review of the literature showed that the cartilage in SC forms in a metaplastic manner in a chronically inflamed synovial membrane and does not represent a neoplastic process.10Y12 However, some authors have reported malignant transformation of the cartilage with development of chondrosarcoma.13

Surgery is the treatment of choice and is the only one that allows an adequate approach to the articular space.

BRIEF CLINICAL NOTES /Mandrioli et al

Fig 2 The right temporomandibular joint of the patient is exposed through a preauricular approach.

Fig 3 Loose bodies.

Fig 4 The postoperative magnetic resonance image in the same patient shows normal findings and no recurrence.

REFERENCES

1. Axhausen G. Pathologie und Therapie des Kiefergelenks. Fortschr Zahnheilk 1933;9:171

2. Von Lindern JJ, Theuerkauf I, Niederhagen B, et al. Synovial chondromatosis of the temporomandibular joint: clinical, diagnostic, and histomorphologic findings. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:31Y38

3. Keogh CF, Torreggiani WC, Munk PL. Bilateral synovial chondromatosis of the temporomandibular joint. Clin Radiol 2002;57:862

4. Nonnan JE, Stevenson ARL, Painter DM, et al. Synovial osteochondromatosis of the temporomandibular joint. An historical review with presentation of 3 cases. J Cranioma- xillofac Surg 1988;16:212Y220

5. Koyama JI, Ito J, Hayashi T, et al. Synovial chondromatosis of the temporomandibular joint complicated by disk displace- ment and calcification of the articular disk: report of two cases. AJNR Am J Neuroradiol 2001;22:1203Y1206

6. Blankenstijn J, Panders AK, Vermey A, et al. Synovial chon- dromatosis of the temporo-mandibular joint. Report of three cases and a review of the literature. Cancer 1985;55:479Y485 7. Holmlund AB, Eriksson L, Reinholt FP. Synovial chondroma-

tosis of the temporomandibular joint. Clinical, surgical and histological aspects. Int J Oral Maxillofac Surg 2003;32:143Y147 8. Petito AR, Bennett J, Assael LA, et al. Synovial chondromatosis of the temporomandibular joint: varying presentation in 4 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90:758Y764

9. Sato J, Suzuki T, Yoshitake Y, et al. The expression of fibroblast growth factor-2 and fibroblast growth factor receptor-1 in chondrocytes in synovial chondromatosis of the temporoman- dibular joint. Report of two cases. Int J Oral Maxillofac Surg 2002;31:532Y536

10. Gynter GW, Dijgraaf LC, Reinholt FP, et al. Synovial inflam- mation in arthroscopically obtained biopsy specimens from the temporomandibular joint: a review of the literature and a proposed histologic grading system. J Oral Maxillofac Surg 1998;56:1281Y1286

11. Milgram JW. The classification of loose bodies in human joints. Clin Orthop 1997;124:282Y291

12. Milgram JW. Synovial chondromatosis: a histopathologic study of thirty cases. J Bone Joint Surg Am 1997;59:792Y801 13. Perry BE, Mc Qeen DA, Lin JJ. Synovial chondromatosis with

malignant degeneration to chondrosarcoma. J Bone Joint Surg Am 1988;70:1259Y1261

Overgrowth of a Costochondral

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