Case Report

Chondrosarcoma of the temporomandibular

joint: a case report and review of the literature
Kyu-Young Oh, Hye-Jung Yoon, Jae-Il Lee, Sam-Pyo Hong, Seong-Doo Hong
Department of Oral Pathology, School of Dentistry and Dental Research Institute, Seoul National University,
Republic of Korea

Objective: Chondrosarcoma is the second most common sarcoma arising in the bone, but it rarely involves
the temporomandibular joint (TMJ). To date, 30 cases of TMJ chondrosarcoma have been reported in the
English literature, and the authors report an additional case arising from a cystic lesion in a 60-year-old
female patient.
Clinical presentation: The clinical and radiological diagnosis of the lesion was initially synovial cyst, and
periodic check-ups were done after aspiration of the lesion. After 3 years, the patient perceived swelling
of the lesion, and surgical excision was performed. The final diagnosis was Grade I chondrosarcoma,
and further immunohistochemical examination was carried out with S-100 protein, D2-40, CD68, and Ki-67.
Conclusion: When clinicians detect a cystic lesion in the radiographic imaging of the TMJ,
chondrosarcoma should be included in the differential diagnosis. In addition, computed tomography
(CT) as well as magnetic resonance imaging (MRI) is recommended for the accurate diagnosis and
proper preoperative planning in TMJ chondrosarcoma. Lastly, further studies on immunohistochemical
examination would be helpful for differential diagnosis in the case of small biopsy in the TMJ.
Keywords: Chondrosarcoma, Temporomandibular joint, TMJ, Immunohistochemistry, Secondary chondrosarcoma

Introduction benign lesion. In addition, only a few immunohisto-

Chondrosarcoma is a malignant tumor composed chemical studies have been carried out in TMJ
entirely of a hyaline cartilage matrix and chondrocytes chondrosarcoma, so the results of additional immuno-
in lacunae without osteoid formation.1,2 Chondro- histochemical examination will be dealt with. To date,
sarcoma constitutes approximately 11% of all primary 30 cases of TMJ chondrosarcoma have been reported
malignant bone tumors and is the second most in the English literature, and they will be summarized
common sarcoma arising in the bone, following osteo- and discussed in this review.
sarcoma.1 More than two-thirds of chondrosarcoma
cases involve the pelvis, shoulder girdles, and the
upper ends of the femur and humerus.2 Chondro- Case Report
sarcoma in the head and neck has been reported to A 60-year-old woman presented with limited mouth
be rare, ranging from 1 to 12% of all cases of chondro- opening due to pain in the right TMJ. Magnetic reso-
sarcoma.1 In the head and neck, the most common sites nance imaging (MRI) showed a cystic lesion in the
of occurrence are the larynx, followed by the mandible, right infratemporal fossa. The clinical diagnosis was
maxilla, and maxillofacial skeleton.3 synovial cyst in the right TMJ. An aspiration was per-
This report presents a case of chondrosarcoma in formed through the sigmoid notch, and the aspirated
the temporomandibular joint (TMJ) arising from a fluid was suggestive of synovial fluid. Two months
cystic lesion. In TMJ cases, there were three reports later, the patient presented suddenly, complaining of
of malignant transformation of benign lesions into pain and paresthesia of the right TMJ. An MRI scan
chondrosarcoma,4–6 but a cystic lesion such as a was performed again and revealed cystic dilatation
synovial cyst has not been reported to be a preexisting with mild synovial enhancement (Fig. 1). Considering
these radiological features, the differential diagnosis
included synovial cyst and pigmented villonodular syno-
Correspondence to: Seong-Doo Hong, Department of Oral Pathology, vitis, which was less likely. The patient refused to
School of Dentistry, Seoul National University, Daehak-ro 101, Jongno-gu,
Seoul, Republic of Korea. Email: hongsd@snu.ac.kr undergo surgery, so periodic check-ups were carried out.

ß W. S. Maney & Son Ltd 2015

DOI 10.1179/2151090315Y.0000000016 CRANIOt: The Journal of Craniomandibular & Sleep Practice 2015 VOL . 00 NO . 0 1
Oh et al. Case report of TMJ chondrosarcoma

Figure 1 Magnetic resonance imaging (MRI) taken 2 months after the first visit. (A) Axial gadolinium-enhanced T1-weighted
MRI showed that the right condyle was surrounded by a cystic lesion with mild synovial enhancement. (B) Axial T2-weighted
MRI showed high signal intensity.

At her 3-year check-up, the patient perceived Surgical excision was performed with partial mandi-
swelling of the right TMJ. On the third MRI, there bulectomy, including the condyle and coronoid pro-
was an increase in size of the cystic lesion, with the cess, after an osteotomy of the right zygomatic arch
greatest diameter being 5.3 cm, but destruction of for surgical approach. During the surgical procedure,
the condyle was not conspicuous (Fig. 2). A punch involvement of the articular fossa was confirmed by
biopsy was taken through the masticator space, and frozen-section examination, and further resection
the result was a hyperplastic cartilage chip, with the was done, including the adjacent dura mater. Sub-
recommendation for excision of the entire mass. sequently, intermaxillary fixation was done, and the
A computed tomography (CT) taken after the separated zygomatic arch was repositioned and fixed
punch biopsy showed a lobulated and low attenuated with a metal plate and screws. For reconstruction, an
mass surrounding the right condyle. The mass had a artificial fossa prosthesis made of ultra-high molecular
peripheral enhancing rim and internal septa, and weight polyethylene was fixed on the glenoid fossa, and
periosteal reaction was detected. Destruction of the a plate with an artificial condyle head was fixed into the
right condyle and erosion of the adjacent cranial mandibular angle. In addition, the latissimus dorsal
base were also found (Fig. 3). flap was used for soft tissue reconstruction.

Figure 2 MRI taken 3 years after the first visit. (A) Axial gadolinium-enhanced T1-weighted MR imaging. A remarkable
increase in size of the cystic lesion was found compared with Fig. 1A. Most of the masticator, retroantral, and parotid spaces
were filled with the lesion. (B) Axial T2-weighted MR imaging. Most portions of the lesion showed high signal intensity.

2 CRANIOt: The Journal of Craniomandibular & Sleep Practice 2015 VOL . 00 NO . 0

 Oh et al. Case report of TMJ chondrosarcoma

Figure 3 Computed tomography (CT) taken after punch biopsy. (A) Axial and (B) coronal CT scans with bone setting pre-
sented prominent condylar resorption (arrows) and erosion of the adjacent cranial base (arrow head). (C) Axial and (D) coro-
nal CT scans with soft tissue setting revealed an enhancing peripheral rim (white arrows) and internal septa (black arrows).

Histopathologic examination of the excised mass Ki-67 were positive in 5% and v1%, respectively.
primarily showed lacunar formation within the chon- The final diagnosis was Grade I chondrosarcoma.
droid matrix (Fig. 4A). Chondrocytes in the lacunae Because involvement of the superior margin was
presented with variation in size and shape, a few of confirmed in the histopathologic examination, the
which were double nucleated (Fig. 4B). Occasionally, patient received 54 Gy radiotherapy in 30 fractions.
two chondrocytes occupied one lacuna. Mitotic There was no evidence of recurrence or distant
figures were extremely rare. The lesion grew in metastasis on CT and positron emission tomography
lobules of various sizes, some of which coalesced, (PET) scans at an 8-month follow-up visit (Fig. 5).
and the lobules were separated by thin fibrous
tissue septa (Fig. 4C). Neither necrosis nor calcifica- Discussion
tion was found within the chondroid matrix. From 1954 to the present, 30 cases of TMJ chondro-
Permeation into the medullary bone of the articular sarcoma have been reported in the English literature
fossa was detected (Fig. 4D). In addition, immuno- (Table 1).3–29 The age ranged from 7 to 75 years old,
histochemical examination was carried out with anti- with the mean age of 46.5 years. The mean age of
bodies against S-100 protein (Clone 4C4.9, 1:300, chondrosarcoma in TMJ was slightly higher than
Cell marque), D2-40 (Clone D2-40, 1:200, Dako), that in the head and neck.30,31 A female predomi-
CD68 (Clone PG-M1, 1:100, Dako), and Ki-67 nance, with a female-to-male ratio of 1.8:1, was
(Clone MIB-1, 1:400, Dako). S-100 was strongly found, which was not consistent with a male
expressed in 80% of the chondrocytes, whereas predominance in the head and neck or other body
D2-40 was weakly expressed in 90%. CD68 and parts.30–32

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Oh et al. Case report of TMJ chondrosarcoma

Figure 4 Histopathologic features of temporomandibular joint (TMJ) chondrosarcoma. Hematoxylin and eosin stains. (A)
Lobular growth pattern of numerous lacunae within a chondroid matrix ( 3 100). (B) Chondrocytes showed marked
pleomorphism, and a double-nucleated chondrocyte was found (arrow, 3 400). (C) Fibrous tissue septa separating coalesced
lobules ( 3 200). (D) Tumor invasion into the medullary bone ( 3 200).

chondrosarcoma. In their case report, hypercellular

content of the lesion and the presence of a chondroid
or myxoid matrix were confirmed through MRI.
In addition, MRI was recommended for preoperative
planning because it provided detailed information on
the anatomic limits and the most accurate view of the
magnitude of the lesion.22,24 In the present case,
however, condylar destruction, erosion of the cranial
base, and mass architecture, including an enhancing
Figure 5 Panoramic radiograph at an 8-month posto- peripheral rim and internal septa, were more
perative follow-up. distinctly found on CT than MRI. Given that these
radiological findings could be clear bases suggesting
Clinically, swelling was the most frequent chondrosarcoma rather than synovial cyst, CT as
symptom of TMJ chondrosarcoma, followed by well as MRI is recommended for the accurate
pain, trismus, and hearing loss (Fig. 6). Headache, diagnosis and proper preoperative planning in TMJ
facial asymmetry, and paresthesia were each reported chondrosarcoma.
once. The major radiological features of TMJ The time from the first perception of symptoms to
chondrosarcoma were condylar resorption, erosion the initial visit to the clinic ranged from 2 to
of adjacent bone, and calcification within the mass 96 months in 26 cases. The mean time of
(Fig. 7). In three reported cases, more destructive 20.7 months in TMJ chondrosarcoma, approximately
bony change than erosion was detected in the three times longer than that in head and neck chon-
external auditory canal, petrous temporal bone, and drosarcoma,31 suggested the propensity for slow
mandibular ramus.3,19,27 An enhancing peripheral growth of the lesion, allowing for its malignancy,
rim was found in four cases and a periosteal reaction and should be considered by clinicians so as not to
in two cases. Oliveira et al.22 mentioned the import- miss chondrosarcoma in the differential diagnosis.
ance of MRI in the differential diagnosis of TMJ In 22 reported cases, the greatest diameter of TMJ

4 CRANIOt: The Journal of Craniomandibular & Sleep Practice 2015 VOL . 00 NO . 0

 Table 1 Thirty cases of temporomandibular joint (TMJ) chondrosarcoma reported in the English literature and the present case

Duration
of
Case symptoms Tumor size Treatment
First author No. Age Sex (months) Chief complaints Radiological features (cm) Grade surgery Follow-up

Gingrass 1 46 F 12 Swelling, pain, Bone deposition – – Surgery –

(1954)7 trismus
Lanier (1971)4a 2 48 F 24 Swelling, pain Condylar resorption 2.0 – Surgery 14 months
Richter (1974)8 3 75 M 10 Swelling, pain, TMJ space widening, elongated condyle, adjacent bony 6.0|5.0|3.5 – Surgery 12 months
hearing loss erosion
Tullio (1974)9 4 17 F 8 Swelling Condylar resorption – – Surgery –
Nortjé (1976)10 5 40 M 6 Swelling, pain TMJ space widening, elongated condyle, radiopacity with 6.0 I Surgery 24 months
radiolucency, adjacent bony erosion
Sato (1977)11 6 – – 36 Pain – – – Surgery, RT –
7 – – 18 Swelling, pain, Radiopacity with radiolucency – – Surgery –
trismus
8 – – 4 Swelling, pain, Radiopacity with radiolucency – – Surgery –
trismus
Cadenat 9 60 F – Swelling, pain – – – Surgery 4 months
(1979)12
Morris (1987)13 10 29 F 24 Swelling, Mass with calcification, adjacent bony erosion 2.5 I Surgery, RT 6 months
headache
Wasenko 11 49 F – Swelling, pain Mass with calcification 4.0|2.5 I Surgery –
(1990)14
15
Nitzan (1993) 12 36 F 72 Swelling, pain, TMJ space widening, condylar resorption, adjacent bony 2.5 I Surgery 7 years
trismus erosion
Merrill 13 50 F 36 Trismus Radiopacity with radiolucency 4.0 – Surgery 18 months
(1997)16b
Sesenna 14 60 F 12 Swelling Mass with calcification – I Surgery 5 years
(1997)17
Ichikawa 15 66 F 12 Swelling, pain, Mass with calcification 2.2|1.5|0.7, – Surgery 3 years
(1998)18b trismus 1.3|1.0|0.5
Batra (1999)19 16 65 M 18 Swelling, hearing Mass with calcification, adjacent bony erosion and 4.0|1.8 I Surgery 7 months
loss destruction
Mostafapour 17 31 F 96 Swelling Condylar resorption 6.0|6.0 I Surgery –
(2000)3

CRANIOt: The Journal of Craniomandibular & Sleep Practice

Oh et al.

18 52 F 18 Swelling Adjacent bony destruction 6.0|4.0|3.0 I Surgery, RT 12 months,

after recurrence
recurrence

2015
Bernasconi 19 55 F 48 Pain, trismus Mass with enhancing peripheral rim, adjacent bony erosion 3.5 I Surgery 1 year
(2004)20
Gallego 20 54 M 3 Swelling, pain, Mass adjacent to condyle 2.2|1.3|0.5 I Surgery 16 months

VOL .
(2009)21 trismus

00
Oliveira 21 11 F 2 Pain, trismus Mass with enhancing peripheral rim, condylar resorption 2.7|1.9 I Surgery 3.5 years
(2009)22
Garzino-Demo 22 65 F 3 Swelling, pain Mass with calcification, condylar resorption – I Surgery, RT 9 years

NO .
(2010)23

0
Case report of TMJ chondrosarcoma

5
6
Oh et al.

Table 1 Continued
Case report of TMJ chondrosarcoma

Duration
of
Case symptoms Tumor size Treatment
First author No. Age Sex (months) Chief complaints Radiological features (cm) Grade surgery Follow-up

González- 23 57 M 12 Swelling, pain Mass with enhancing peripheral rim, condylar resorption – I Surgery 2 years
Pérez (2011)24
Xu (2011)5c 24 34 F – Trismus, facial Mass with calcification, condylar resorption, adjacent bony 8.0|6.0|5.0 I Surgery –

CRANIOt: The Journal of Craniomandibular & Sleep Practice

asymmetry erosion
Ramos- 25 45 M – Asymptomatic Multilocular radiolucency with mandibular osteolysis 5.5|2.0 II Surgery 3 years
Murguialday

2015
(2012)25
Abu-Serriah 26 48 M 2 Pain Mass with calcification, adjacent bony erosion 1.0 – Surgery 6 months
(2013)26

VOL .
Goutzanis 27 23 M 2 Swelling, pain Condylar resorption, adjacent bony destruction 6.0 I Surgery 2 years

00
(2013)27
Coleman 28 63 F – Swelling Lobulated chondroid mass, condylar resorption 5.5|5.0|4.0 – Surgery, RT –
(2013)6d

NO .
0
Giorgione 29 56 M 12 Swelling, pain, Mass with calcification, condylar resorption, periosteal 4.6|4.0|3.9 I Surgery, RT –
(2013)28 trismus reaction
Kumar Reddy 30 7 M 12 Swelling, pain Mass with calcification, condylar resorption 5.0|3.0 I Surgery 1 year
(2014)29
Present case 31 60 F 36 Swelling, pain, Lobulated mass with enhancing peripheral rim and internal 5.3 I Surgery, RT 5 months
(2014) trismus, septa, condylar resorption, adjacent bony erosion, periosteal
paresthesia reaction
 Oh et al. Case report of TMJ chondrosarcoma

Figure 6 Chief complaints of 28 cases of TMJ chondrosarcoma in the English language literature.

Figure 7 Radiological features of 29 cases of TMJ chondrosarcoma in the English language literature.

chondrosarcoma ranged from 1.0 to 8.0 cm, with the that of solitary osteochondroma was 0.4–2%.35–37
mean size of 4.3 cm. The tumor size of chondrosar- In reports of TMJ chondrosarcoma, three cases of
coma in 2TMJ was similar to that in the skull base secondary chondrosarcoma arose from synovial
but smaller than that in the head and neck.31,33 chondromatosis, osteochondroma, and enchon-
Temporomandibular joint chondrosarcoma has droma, respectively.4–6 In the present case, the initial
typical histopathologic features of chondrosarcoma diagnosis was a synovial cyst, based on the MRI
in that lacunae, including pleomorphic chondrocytes, scan. Although the diagnosis was not definitive
show a lobular growth pattern within the chondroid because a biopsy was not performed at that time,
matrix, and double-nucleated nuclei are occasionally synovial cyst was the most probable diagnosis,
detected. According to Evans et al.,32 chondrosar- based on the images showing the cystic lesion with
coma has been classified into three grades on the synovial enhancement and the fluid from the aspira-
basis of histologic criteria, including nuclear size, tion. As treatment for a synovial cyst, an arthro-
mitosis, cellularity, and matrix character. In this scopic procedure instead of surgical excision was
review on histologic grading of TMJ chondrosar- suggested for its conservativeness and management
coma, by classifying ‘well-differentiated’ or ‘low of inflammation.38 However, if the cyst does not
grade’ into ‘Grade I’, 18 out of 19 cases were disappear spontaneously after the arthroscopic pro-
diagnosed as Grade I, with only one as Grade II. cedure, surgical excision should be carried out to pre-
This demonstrated that the percentage of Grade I is vent aggravation of the lesion. Although malignant
much higher in TMJ chondrosarcoma than in other transformation of synovial cysts has not been
body parts.32,34 reported to date, clinicians are advised to notice the
Malignant transformation from a benign lesion, possibility of malignant transformation, taking the
including synovial chondromatosis, multiple chon- present case into account.
droma, or osteochondroma, to chondrosarcoma has Out of 31 cases of TMJ chondrosarcoma, fine
been reported as secondary chondrosarcoma.35 needle aspiration biopsy (FNAB) was performed in
In previous studies, the malignant transformation eight cases. Among these cases, the diagnosis of
rate in synovial chondromatosis was 5–10% and chondrosarcoma could be inferred from FNAB in

CRANIOt: The Journal of Craniomandibular & Sleep Practice 2015 VOL . 00 NO . 0 7

Oh et al. Case report of TMJ chondrosarcoma

only two cases; in one of these cases, the second case declined radiotherapy despite positive margins
FNAB was necessary for diagnosis due to inade- in the histopathologic analysis. Considering its
quacy of the first biopsy.19,23 In four cases, FNAB efficacy as mentioned above, postoperative radiother-
was not indicative of chondrosarcoma20,24,27 or insuf- apy should be applied to cases of TMJ chondrosar-
ficient for definitive diagnosis.13 Two of the cases coma with incomplete surgical resection margin.
reported that the result of FNAB was pleomorphic
adenoma.17,28 In the present case, a small punch Disclaimer Statements
biopsy was performed, and the result implied carti-
Contributors
lage proliferation but was not sufficient for definitive
K-YO, as a first author, contributed to selecting the case
diagnosis as chondrosarcoma. A small amount of
and writing the article in whole. H-JY, J-IL, and S-PH
tissue from FNAB or a punch biopsy in chondro-
contributed in reviewing the literature and revising the
sarcoma shows pleomorphic chondrocytes and
article in part. S-DH, as a corresponding author, con-
double-nucleated nuclei, but these cellular features
tributed in directing all the process and revising finally.
can also be seen in other chondroid lesions like syno-
vial chondromatosis.39 Funding
To take advantage of FNAB in TMJ lesions suspi-
cious for chondrosarcoma, immunohistochemical Conflicts of interest
stains may play a major role in reaching a reliable There are no conflicts-of-interest.
diagnosis. To date, only six cases of TMJ chondro-
sarcoma had immunostaining.17,18,21,22,24,28 In the Ethics approval
results, S-100 was positively expressed in 5–35% of Not required. Because this is a report of a single case
chondrocytes17,21,24 and Ki-67 was in 5–50%.21,24,28 not including private information such as patient’s
Compared to these results, the present case showed identification and face photos.
high expression of S-100 and low expression of
Ki-67. Additionally, D2-40 and CD68 immunostains References
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