Dentomaxillofacial Radiology (2008) 37, 213219 2008 The British Institute of Radiology http://dmfr.birjournals.

org

RESEARCH

An investigation of magnetic resonance imaging features in 14 patients with synovial chondromatosis of the temporomandibular joint
M Ida*,1, H Yoshitake2, K Okoch1, A Tetsumura1, N Ohbayashi1, T Amagasa2, K Omura3, N Okada4 and T Kurabayashi1
1 Oral and Maxillofacial Radiology, Division of Oral Health Sciences, Graduate School, Tokyo Medical and Dental University, Japan; 2Maxillofacial Surgery, Division of Oral Health Sciences, Graduate School, Tokyo Medical and Dental University, Japan; 3 Oral and Maxillofacial Surgery, Division of Oral Health Sciences, Graduate School, Tokyo Medical and Dental University, Japan; 4 Diagnostic Oral Pathology, Division of Oral Health Sciences, Graduate School, Tokyo Medical and Dental University, Japan

Objectives: To show the characteristic MRI features of synovial chondromatosis (SC) of the temporomandibular joint (TMJ). Methods: All patients with histologically proven SC of the TMJ who underwent MRI at our clinic were examined. In 14 patients (male-to-female ratio, 2:12; average age 4614 years), clinical and conventional radiographic findings were reviewed. In addition, the MRI findings of articular disc and condyle position, shape and signal intensity of the joint spaces, and bone changes of surrounding structures were analysed. Results: The main symptoms were pain (in 93% of the patients) and limitation of mouth opening (64%). Two cases showed typical multiple calcifications around the TMJ on conventional radiography. On MRI, the disc position was normal in 12 (86%) patients and the condyle was inferiorly displaced in 9 (64%) patients. 11 (79%) patients showed enlargement of the joint space, with either a dumbbell shape or bulging. SC in the upper compartment showed various degrees of bone changes of the articular eminence and fossa. SC in the lower compartment showed concavity or hypertrophy of the condyle. The severity of the bone changes progressed with duration of symptoms. Conclusions: About 0.3% of the patients complaining of TMJ pain and dysfunction were found to have SC. There was great variation in the MRI features of the TMJs with SC. More severe destruction of surrounding bone structures with features resembling a tumour were found in patients with a longer duration of symptoms. Dentomaxillofacial Radiology (2008) 37, 213219. doi: 10.1259/dmfr/95185114 Keywords: magnetic resonance imaging; chondromatosis; synovial; temporomandibular joint

Introduction Synovial chondromatosis (SC) is a joint disease that affects mainly large joints such as the knee, elbow or hip.1 The aetiology is not clear. SC of the temporomandibular joint (TMJ) is thought to be very rare compared with the frequency of occurrence in large joints. The symptoms of SC of the TMJ are not
*Correspondence to: Dr Mizue Ida, Oral and Maxillofacial Radiology, Division of Oral Health Sciences, Graduate School, Tokyo Medical and Dental University, 5-45 Yushima 1-chome, Bunkyo-ku, Tokyo 1138549, Japan; E-mail: ida.orad@tmd.ac.jp Received 13 December 2006; revised 31 May 2007; accepted 31 May 2007

characteristic. Many patients present with temporomandibular dysfunction (TMD).2,3 Some cases have been misinterpreted as a tumour of the parotid gland.4,5 Therefore, the primary diagnosis of SC of the TMJ had been thought to be rare.2 Conservative surgical treatment is recommended for SC.5,6 However, several cases that showed invasive features have been reported.710 On surgically excised specimens, SC has sometimes been diagnosed as chondrosarcoma.1,11 Therefore, presurgical imaging is important. Detecting SC of TMJs by conventional X-ray imaging appears to depend on the amount of calcification of loose

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Table 1

TMJ synovial chondromatosis M Ida et al

Clinical findings of patients with synovial chondromatosis of the temporomandibular joint (TMJ) Side R L L R L R R R L R R L R L Duration{ Compartment* (months) U L U U U U U L U U U&L L U&L U 12 48 1 12 48 8 16 360 2 12 48 36 60 7 Symptoms{ PN, SD, DEV LO, PN LO, SW, PN LO, PN LO, PN, SD LO, PN, SD, DEV LO, PN, SD LO, SW, open bite PN, DEV PN LO, PN, SD LO, PN, SD LO, PN PN, SD TMD treatment Yes No Yes Yes No Yes Yes Yes Yes Yes Yes Yes Yes No Histopathological diagnosis Synovial chondromatosis Synovial osteochondromatosis of the left TMJ Synovial chondromatosis with CPPD deposition of the left TMJ Synovial chondromatosis Synovial chondromatosis of the left TMJ Synovial chondromatosis Synovial chondromatosis of the right TMJ Synovial osteochondromatosis of right TMJ Synovial Synovial Synovial Synovial TMJ Synovial Synovial osteochondromatosis chondromatosis chondromatosis of the TMJ osteochondromatosis of the left chondromatosis of the right TMJ chondromatosis of the left TMJ

Age Case No. Gender (years) 1 2 3 4 5 6 7 8 9 10 11 12 13 14 F F F M F F F F F F M F F F 23 25 45 50 38 57 39 64 56 46 71 33 59 36

*Affected compartment of the joint at surgery. {Duration of the symptoms before MRI. {Symptoms at MR examination. TMD, temporomandibular disorder; F, female; M, male; R, right; L, left; U, upper; L, lower; PN, pain; SD, sound; DEV, deviation of chin at mouth opening; LO, limitation of mouth opening (intercisal distance ,40 mm); SW, swelling; CPPD, calcium pyrophosphate dihydrate

bodies. Similarly, detectability by CT examination also depends on the extent of calcification of the loose bodies12,13 although more cases were detected with CT than by conventional radiography.14,15 Arthroscopy seems to be one of the best modalities for detecting intracapsular non-calcified free bodies.1517 However, it may not be the first choice for a patient with TMJ discomfort because of the invasive nature of the procedure. MRI has recently been widely used to image the TMJ because of its ability to visualize soft tissues. Several papers have reported MRI characteristics of SC of the TMJ.5,18 However, differentiation of SC from tumours by imaging may be difficult, especially for cases that show evidence of bone destruction or invasion. We reviewed all of our patients with SC who underwent MRI of the TMJ and analysed the images to determine the characteristic MRI findings of SC.

3 inch surface coil. Pulse sequences were as follows: (1) sagittal closed-mouth spin echo (SE) T1 weighted image (T1WI); (2) sagittal closed-mouth turbo-SE (TSE) T2 weighted image with fat-suppression pulse (T2WI); (3) coronal closed-mouth SE T1WI, (4) sagittal openmouth TSE T1WI. A rectangular field-of-view of 906120 mm was applied. Slice thickness was 3 mm with an interslice gap of 0.3 mm. In addition, gadolinium-enhanced T1 weighted images were obtained from two patients. All of the images of patients with SC were reviewed.

Results The 15 patients with SC of the TMJ who had MRI constituted 0.3% of the 4700 patients who underwent MRI examination of TMJs during the period indicated above. One patient was excluded from the analysis because her joint had co-existing pigmented villonodular synovitis. 13 of the remaining 14 patients were diagnosed with primary SC and the remaining patient was diagnosed with secondary SC provoked by calcium pyrophosphate dihydrate (CPPD) crystal deposition. The most common complaints of the TMJ region at the first visit to our hospital were pain in ten patients, limitation of mouth opening in eight patients, swelling in two patients and/or occlusal change in two patients. The patients are listed in Table 1. Male-to-female ratio was 2:12. The average age was 4614 years with a range of 23 years to 71 years. The right joint was affected in eight patients and the left in six patients. At surgery, 11 joints were found to be affected in the upper compartment of the joint space and 2 of those joints

Materials and methods From August 1995 to July 2006, all patients at our institution who were surgically and histopathologically proven to have SC of the TMJ were reviewed. 16 patients were diagnosed with SC of the TMJ and 15 of them underwent MRI prior to surgery. The total number of patients who underwent MRI of the TMJs because of TMD and/or TMJ pain at our institution during this time was 4700. All patients had panoramic radiographs, and transcranial and transorbital radiographs of both TMJs before MRI. MRI was performed on a 1.5 T scanner (Magnetom Vision; Siemens AG, Erlangen, Germany) with a dual
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showed extension of the disease to the lower compartment. The remaining three joints were affected only in the lower compartment. The duration of symptoms before MRI was 1 month to 30 years (average 4.3 years, median 1 year). Ten (71%) patients received various treatments for TMD before MRI, such as non-steroidal anti-inflammatory drugs, occlusal splints and/or self-mobilization exercises. Two (14%) patients experienced transient alleviation of symptoms after treatment. At the time of MRI, 13 (93%) patients had pain on mouth opening. Clinical signs included limitation of mouth opening in nine (64%) patients, joint sounds in seven (50%) patients, occlusal change in four (29%) patients and swelling in the TMJ region in two (14%) patients. Conventional radiographs showed calcification around the TMJ (Figure 1a) in two (14%) cases, enlargement of the joint space in eight (57%) cases, and erosion or deformation of bone in seven (50%) cases. No abnormality was detected by conventional radiography in two (14%) cases. MRI findings of the TMJ with SC are shown in Table 2, in which patients are aligned in order of the duration of the symptoms and affected compartment of the joint space. The disc position was normal in 12 (86%) cases and 2 showed anterior displacement of the disc. Nine (64%) patients showed inferior displacement or subluxation of the condyle. 11 (79%) cases out of 14 showed bulging of the joint space. Eight showed a characteristic dumbbell

shape bulging either anteroposteriorly or mediolaterally, being constricted at the top of the condyle (Figure 1b). All 11 enlarged joints, dumbbell shaped or bulging, showed signal intensity slightly higher than or equal to muscle on T1WI. On T2WI, 9 joints showed effusion-like high signal intensity containing multiple signal void particles or lines (Figures 1 and 2). Two joints showed intermediate or mixed signal intensity with a small amount of effusion (Figure 3). Three patients had no evidence of capsule enlargement. In one patient (case 6), there was effusion in the upper joint compartment with a small number of small signal void particles and lines (Figure 4). MRI diagnosis was felt to be inconclusive, but numerous cartilaginous particles were found within the upper compartment of the right TMJ by arthroscopic examination. 6 of 11 patients with SC of the upper compartment showed bone changes of the eminence and fossa, including flattening, concavity, osteophyte formation, erosion and perforation of the fossa. The average duration of the symptoms in the patients with bony changes was 3.42.3 years and 0.7 0.4 years for those without. The bony changes of the eminence and fossa were more various and severe in the patients with a duration of symptoms of over 1 year. One case with a 4 year history of symptoms showed perforation of the fossa and subdural extension of the lesion (Figure 5). The two patients who had evidence of loss of the compartmentalization between upper and lower joint spaces had the longest duration of symptoms.

Figure 1 Case 1. (a) Cropped panoramic radiograph showing multiple calcifications around the right temporomandibular joint (TMJ). (b) Oblique sagittal T2 weighted MR image of the right TMJ, showing a dumbbell-shaped enlargement of the capsule with effusion and multiple signal-void areas
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Table 2 Signal intensity Disc position Normal ADD woR Normal Normal Normal ADD wR Normal Normal Normal Normal Normal Normal Normal Normal No bulging Bulging (A) No bulging 5M .5M 5M High+ low signal grains High+a grain High+grainy low signals None None None Bulging (A) 5M Heterogeneous Dumbbell (AP) 5M High+cotton wool Dumbbell (AP) .5M 5 M+signal void grains No bulging Dumbbell (AP, LM) Dumbbell (AP) Dumbbell (AP) Dumbbell (AP, LM) 5M .5M .5M .5M .5M High+lines High+signal void grains High+signal void grains High+signal void Grains High+cotton wool None Flattening None None Erosion, thinning of fossa Erosion, flattening Bulging (P) Dumbbell (AP) Dumbbell (AP,LM) .5M 5M 5M High+sandy signal void High+cotton wool High +cotton wool None Erosion None None None None Capsule T1WI T2WI Fossa Tubercle Condyle Bone changes

MRI findings of synovial chondromatosis of the temporomandibular joint (TMJ) (in order of symptoms and compartment)

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No.

Duration (months)

Compartment

Condyle position

3 9 14

1 2 7

U U U

Anterior Lateroinferior Normal

6 1 4 10 7

8 12 12 12 16

U U U U U

Normal Anterior Anteroinferior Normal Anteroinferior

None Flattening Osteosclerosis pseudocyst None None None Concavity None None

48

Anterior

11

48

U&L

Subluxation

13

60

U&L

Normal

Erosion, expansion, perforation Erosion, osteophyte

None Erosion None None Erosion, osteophyte, Erosion, flattening Erosion, osteophyte None None None None

TMJ synovial chondromatosis M Ida et al

12 2 8

36 48 360

L L L

Inferior Normal Subluxation

Erosion, osteophyte Erosion, flattening Hypertrophy Concavity Hypertrophy

T1/T2 WI, T1/T2 weighted image; U, upper; P, posteriorly; . 5 M, slightly higher than muscle; ADD woR, anterior disc displacement without reduction; AP, anteroposteriorly; 5 M, comparable signal intensity with muscle; LM, lateromedially; ADD wR; anterior disc displacement with reduction; L, lower; A, anteriorly Figure 2 Case 7. Sagittal T2 weighted MR image of the right temporomandibular joint, showing a dumbbell-shaped enlargement of capsule with effusion and cotton wool appearance. At surgery, numerous lightly calcified cartilaginous particles were found in the upper compartment

Figure 3 Case 4. Sagittal T2 weighted MR image demonstrating a dumbbell-shaped enlargement of the capsule with mixed signal intensity. At surgery, elastic soft masses were found in the upper compartment that were histologically confirmed as a proliferation of hyaline cartilagenous nodules embedded in connective tissue

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Discussion The most common symptoms of patients with SC in previous reports were swelling and pain.2,3,18 Those in the present series experienced limitation of mouthopening and pain. Only two of our patients complained of swelling in the TMJ region. Numerous calcified particles around joints are an SC-specific radiographic finding and this was found only in two of our patients. The other radiological findings such as enlargement of the joint space or bone changes are not characteristic of the disease. Consequently, 11 (79%) patients were treated with a clinical diagnosis of TMD. They underwent non-surgical treatment for TMD and did not experience any significant relief of the symptoms. MRI has recently been widely used for TMJ dysfunction syndrome because of its ability to visualize the articular disc. Kim et al18 analysed 11 cases with SC of the TMJ by MRI and reported that SC should be considered when the amount of synovial fluid is abnormally large, disc position is fairly normal and there is no associated severe bone change. Our 14 cases with SC showed more diversified MRI features. Although the majority of patients showed a typical enlargement of the capsule with effusion, some showed an enlargement with heterogeneous signal intensity on T2WI that did not indicate effusion but a space-occupying mass. There were some cases that only showed a small amount of effusion and a few particles without enlargement. When a patient with TMJ pain does not respond to conventional TMD treatment, MRI should be considered and SC included in the differential diagnosis. We had three patients with SC in the lower compartment. Reported cases with SC in the lower compartment

Figure 4 Case 6. Sagittal T2 weighted MR image of the right temporomandibular joint, showing effusion and a small number of signal-void grains and lines. Arthroscopic examination showed numerous non-calcified small free bodies in the upper compartment

Eight patients showed bone changes of the condyle. Five had SC in the lower compartment. Three patients had no SC in the lower compartment but showed concavity, flattening or osteosclerosis of the condyle. Two patients with SC of the lower compartment showed hypertrophic changes of the condyle (Figure 6). The hypertrophic condyle heads were surgically excised and histologically proved as proliferation of cartilaginous and synovial tissue connected to hypertrophic bone.

Figure 5 Case 12. (a) Sagittal T1 weighted MR image (T1WI) of the right temporomandibular joint in the closed-mouth position, showing a mass of intermediate signal intensity superior and posterior to anteriorly displaced condyle. (b) Gadolinium-enhanced coronal T1WI with fat suppression image of the same joint showing subdural invasion (arrow heads) by an enhanced mass
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Figure 6 Case 13. (a) Sagittal T1 weighted images of the left temporomandibular joint showing dislocation of the condyle and hyperplastic elongation of its anterior aspect. (b) Schematic illustration of (a). A small free body was in the anterior pouch of the lower compartment and a larger body was in the posterior joint space. The excised hyperplastic area proved to be composed of a proliferation of cartilaginous and synovial tissue connected to hypertrophic bone. The free bodies were composed of cartilage mixed with bone. EAC, external auditory canal

are scarce.1820 All the cases in the present series showed a relatively small number of signal-void particles within an effusion. Two of them showed hypertrophy of the condyle. The hypertrophic bone was histologically determined to be proliferation of cartilaginous and synovial tissue connected to hypertrophic bone. Similar condylar enlargement has been reported in other cases of SC in the lower compartment.19,20 Hypertrophic deformation of the condyle might indicate a characteristic feature of SC in the lower compartment. We performed enhanced MRI with gadolinium for two patients. One patient showed enhancement of the capsule but no enhancement of its content. This feature is compatible with synovial thickening with a large amount of effusion. Another patient showed diffuse heterogeneous enhancement of the entire area of enlargement. This finding suggested a space-occupying mass in the TMJ. At the time of surgery, the joint space was found to be filled by an elastic hard mass. Histopathological examination showed that this was composed of numerous cartilaginous nodules mixed with synovial tissues. The extent of bone destruction was more severe in patients with longer duration of symptoms. Usually,

the first symptom noticed by patients was pain. If it is assumed that pain occurs when cartilaginous particles become detached from the synovium into the joint space, proliferation, enlargement or aggregation of the particles will progress thereafter. The destruction of bone could be attributed to compressive pressure.3 Although SC is frequently referred to as a nonneoplastic condition, it may consequently cause grave symptoms.9 However, paradoxically, invasive-looking SC of the TMJ is thought to be in the end stage of the disease and have low risk of recurrence after conservative treatment such as simple extirpation.21 In conclusion, 15 patients with SC were found among those who complained of TMJ pain and dysfunction. SC constituted 0.3% of all the MRI cases of TMD. The MRI features of the TMJ with SC were highly diversified. Except for a typical feature of enlargement of the joint capsule by effusion accompanied with signal void particles, some showed mixed signal intensity of the enlargement on T2WI and some did not show enlargement. In cases with a longer duration of symptoms, the surrounding bone was destroyed and appearances suggested the presence of a tumour.

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