Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx

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Journal of Oral and Maxillofacial Surgery, Medicine, and

Pathology
journal homepage: www.elsevier.com/locate/jomsmp

Chondrosarcoma in the condyler head: A case report and review of the

literature
Toru Inomataa,b,*, Kouki Miuraa, Chihiro Fushimia,c, Chihiro Kannoa, Masao Kurosakac,
Chihiro Matsuid
a
Department of Head and Neck Oncology and Surgery, International University of Health and Welfare Mita Hospital, Tokyo, Japan
b
Department of Oral and Maxillofacial Surgery, Nippon Dental University Hospital, Tokyo, Japan
c
Department of Head and Neck Oncology and Surgery, Chiba Tokushukai Hospital, Chiba, Japan
d
Department of Plastic Surgery, Juntendo University Hospital, Tokyo, Japan

A R T I C LE I N FO A B S T R A C T

Keywords: Chondrosarcoma is accounts for some 10 % of osteogenic malignant tumors, its development from a jawbone is
Chondrosarcoma very rare. We have experienced a patient with chondrosarcoma developing in the condyler head as reported
Chondyar head below. The patient was a 42-year-old man. elastic hard swelling was observed in a location corresponding to the
Head and neck left condyler head. Radiographically, a neoplastic lesion was detected in the left head of mandible with no bone
Tumor grade
destruction in the skull base. Histopathological examination was performed under general anesthesia. Based on
the diagnosis of suspected condyler head chondrosarcoma, tumor resection,left supraomohyoid neck dissection
and reconstruction with an anterolateral thigh flap were performed under general anesthesia of the next month.
A histopathologycal observation of removed tissue samples confirmed chondrosa-rcoma. No abnormality such as
tumor recurrence has been found for seven years postope-ratively.

1. Introduction
Although chondrosarcoma accounts for about 10–20 % of primary
bone mal-ignancies, the occurrence from the jaw bone is extremely
rare. We report a case of chondrosarcoma occurring in the condyler
head.
2. Case report
A 42-year-old man was pointed out by a nearby doctor with swelling
of the left condyler head in February 2013, and was referred for ex-
amination and treatment. No abnormal findings were found in any
medical history or general condition. Local findings revealed elastic-
hard swelling in the left condyler head. His face was asymmetric, and
diffuse swelling was noted over an area of approximatery 30 × 35 mm
in the left preauri-cular region. There were no other symptom than
swelling. Mention range of maximum mouth opening before surgery
was 35 mm.
X-ray radiography showed an irregular-shaped radiopaque mass in
the left condylar head. On CT, the border was relatively clear, and the
tumor size was 40 × 30 × 25 mm, enlarged and irregular, solid and
enlarged at the left condyler head, but bone destruction at the skull base
was not observed. Under soft tissue conditions, the inside was solid and
neoplastic lesions with punctate calcification were noted. In addition,
lymph node swelling was noted in the left mandible. MRI revealed an
internal nonuniform solid lesion with low and high signals at T1-WI and
T2-WI images in the left mandible. The surrounding muscles were in a
state of contraction and no surrounding tissue infiltration was observed
(Fig. 1). PET-CT revealed accumulation of SUVmax = 6.5 in the left
condyler head.
The clinical diagnosis was suspicion of the left mandibular condyler
tumor, and in order to obtain a definitive diagnosis, histopathological
examination by anterior incision was performed under general an-
esthesia in April 2013. A flap was raised on the parotid capsule, and
when it reached the tumor, it was partially resected into a spindle and
samples were collected. The histopathological findings indicated that
there is a possibility of chondrosarcoma or mesenchymal chondor-
osarcoma in which calcified spots are scattered and large nuclei are
prominent in cell components.
In May 2013, Under general anesthesia, left hemimandibectomy,
left supraomohyoid neck dissection, and reconstruction using the
anterolateral thigh flap were performed.
Extend the incision line at the time of histopathological examination
to the lower jaw, the facial nerve was preserved, and surgical free

⁎
Corresponding author at: Department of Oral and Maxillofacial Surgery, Nippon Dental University Hospital, 2-3-16 Fujimi,Chiyoda-ku, Tokyo, 102-8158, Japan.
E-mail address: t-ino@tky.ndu.ac.jp (T. Inomata).

https://doi.org/10.1016/j.ajoms.2020.07.002
Received 15 May 2020; Received in revised form 10 July 2020; Accepted 12 July 2020
2212-5558/ © 2020 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd All rights reserved.

Please cite this article as: Toru Inomata, et al., Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology,
https://doi.org/10.1016/j.ajoms.2020.07.002
T. Inomata, et al. Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx

Fig. 1. (a and b) Axial and coronal CT showing Centered on the left mandibular condyle, the border was relatively clear, and a tumor lesion with irregular limbus and
solid swelling punctate calcification was observed, but no bone destruction of the skull base was observed. (c) MRIT1-WI showing an irregular-shaped mass around
the left condylar head that gave low-intensity signals. (d) MRIT2-WI showing an irregular shaped mass around the mandible with isointense and heterogeneous low-
high intensity signals.

margin was 10 mm, the medial pterygoid muscle, lateral pterygoid
muscle around the tumor, and the condyler head including the articular
disc and synovium, coronoid process were resected. The mandibular
fossa drilled out. Because left submandibular lymphadenopathy was
observed, we performed supraomohyoid neck dissection. The excision
site was filled with de-epithelialized anterolateral thigh flap to prevent
dead space, and microvascular anastomosis was performed. End-to-end
anastomosis to thyroid artery and end-to-side anastomosis to internal
jugular vein (Fig. 2). The specimen was a 40 × 35 × 30 mm lobule-like
mass, and the fractured section was a pale yellowish white, solid, car-
tilage-like tissue centered on the condylar head (Fig. 3). Histologically,
in multicentric nodular cartilage tissue, the cell density was not so high,
but in the high place, the size was different in the nucleus, and bi-
nuclear ones were scattered (Fig. 4). The stump was negative and no
metastasis was found in the cervical lymph node. The final histo-
pathological diagnosis was a diagnosis of condylar head GradeⅠchon-
drosarcoma. Mention range of maximum mouth opening immediately
after surgery was 22 mm. Postoperative Seven years, mention range of
maximum mouth opening has recoverd to 35 mm, but no signs of re-
currence, metastasis have been found (Fig. 5).
3. Discussion
Among primary bone malignancies, chondrosarcoma is most fre-
quently followed by osteosarcoma and is considered to be about
10–20% [1,2]. Favorable age is from 30 to 40 years old, and male to
male ratio for occurrence tends to be more in males at 1: 1 to 10: 1. It
occurs anywhere in the bone tissue, and it is said that there are many
long bones and ribs such as the pelvis and femur. Cause disease is
primary disease in patients with Werner syndrome or hereditary mul-
tiple exosteopathia, multiple endochondromatosis such as Ollier disease
and Maffucci disease, secondary disease with bone Paget's disease, It
has been reported that the frequency is low but that it is induced after
irradiation. There are no specific findings in the imaging findings, but it
is depicted as an ambiguous lesion that extends to soft tissues and
partially calcifies [3].
The chondrosarcoma in the head and neck region is 5–10 % of the
total chondrosar-coma and is often found in the nasal sinuses and
larynx, and the mandibular primary is considered to be rare [1,4]. Nasal
sinus lesions are accompanied by nasal congestion, laryngeal lesions are
accompanied by hoarseseness and respiratory distress, but the lower

2
T. Inomata, et al. Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx

Fig. 2. The condyler head including the coronoid process, the joint disc and the synovium was resected. (b) preserving the facial nerve, and the left submandibular
lymph node was enlarged. Arrow is facial nerve. (c) The resected portion was filled with deepithelial anterolateral thigh flap to prevent dead space, and microvessel
anastomosis was performed. Arrow is deepithelial anterolateral thigh flap. Arrowhead is pedicle.

Fig. 3. The specimen was a 40 × 35 × 30 mm lobule-like mass, and the fractured section was a pale yellowish white, solid, cartilage-like tissue centered on the
condyler head (Cutting surface).

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T. Inomata, et al.
Fig. 4. Microphotographs of hematoxylin and eosin-stained sections of man-
diblar tumor. (a) At low magnification, the tumor consists of eosinophilic, pale,
and basophilic areas that irregularly intermingle with each other. (b) In baso-
philic areas higher magnification, multicentric nodular cartilage tissue, the cell
density was not so high, but in the high place, the size was different in the
nucleus, and binuclear ones were scattered.
jaw is often asymptomatic [5]. It may show a bright bone resorption
image. Differential diagnosis in the mandible area includes amelo-
blastoma, chondroma, synovial osteochondromatosis,chondroblastic
osteosarcoma [6–8].
From 1954–2019, 25 cases of chondrosarcoma in the condylar head
were reported in the English literature (Table 1) [3,9–30]. The ages of
these patients, including the present case, ranged from 23 to 78 years
(mean, 47.2 years). These values differ from those cited in reports on
facial skeletal chondrosarcoma, of which the number of male patients
was slightly higher than that of female patients [11,12,31].
The most common symptom of chondrosarcoma in the condyler
head is preauricular swelling(18/25 cases), frequent pain(13/25 cases),
and mildly or severely limited mouth opening(8/25 cases). Hearing loss
was reported in three cases, with case 3 [11]being due to pressure from
tumor on the audinary tube, resulting in otitis media, and cases 6 and
10 [14,18]being due to the occulusive effect of the tumor on the ear
canal.
4
Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx
Cohen et al. [32] stated that low-grade chondrosarcomas, which are
often difficult to diagnose on plain radiography and to differentiate
from enchondromas, are characterized by the MR tandem of low signal
intensity septa on T2-WI together with septal or ring-and-arc en-
hancement. In the present case, resorption of the mandibular condyle
and an irregular-shaped mass with calcification were apparent on
imaging. MRI showed an irregular-shaped mass that gave low-intensity
signals on T1-WI and heterogeneous low-high intensity signals on T2-
WI. The mass-like shadow was enhanced, but an unclear broundary was
enhancedin the surrounding soft tissue. Histological examination of the
chondorosarcoma revealed proliferation of hyaline cartilage with sar-
comatous stroma containing stellate, spindle-shaped, or rounded cells.
O’Neal [33] and Evans et al. [34] established classifications to
evaluate the malignancy of chondorosarcoma. Evans et al. [34] classi-
fied chondrosarocoma into three degrees
Based on nuclearsize, nuclear staining, frequency of mitosis, and
cellularity. GradeⅠ tumors are moderately cellular and contain hyper-
chromatic and plump nuclei of uniform size, with binucleated cells
occasionally observed and mitosis not observed.GradeⅡ
tumorsare more cellular and contain a significant proportion of
moderately-sized nuclei, with mitosis occasionally observed.
GradeⅢtumors are more cellular, pleomorphic, and atypical than
GradeⅡ, with mitosis more frequently observed than in lower grades.
Based on this approach, we classified our case of chondrosarcoma of
the left condylar head as GradeⅠ.
Local reccurrence is high regardless of grade, Saito et al. [35], Re-
ported that local recurrence rate was 33.3 % among 56 cases of chon-
drosarcoma in the head and neck region, and Ruark et al. [36], Re-
ported that recurrence rate was 88 %. Although distant metastasis is
rare, the most frequent metastatic site is the lung, followed by lymph
nodes, sternum, and vertebrae. The relationship between histo-
pathology and tumor behavior was examined in 71 cases of chon-
drosarcoma [34].The 5-year survival rates for each grade were as fol-
lows:GradeⅠ(90 %), Grade Ⅱ(81 %), GradeⅢ(43 %). The 10-year
survival rates for each grade were as follows:GradeⅠ(83 %), GradeⅡ(64
%), and GradeⅢ(29 %).
Metastasis was not observed for any cases with GradeⅠtumors but
was observed for 10 % of those with GradeⅡtumors and 71 % with
GradeⅢtumors. No definitive relation-ship was found between tumor
grade and local recurrence, as local recurrence is primarily dependent
on the adequacy of surgical therapy rather than histological grade.
For the treatment of chondrosarcoma, surgical excision has been
reported as the first choice because of its low radiosensitivity and little
effect on chemotherapy [37]. In the case of the mandible at the primary
jaw, patients with complete resection after initial treatment were alive
with no disease on average for 3 years and 6 months, but Saito et al.
[35], Showed local recurrence after extended resection, but additional
resection reported that they had survived disease-free for 8 years and 3
months, and that Acar et al. [5], reported that local control after ra-
diation therapy could achieve local control for 3 years and 7 months by
performing complete resection. The prognosis was considered to be
good if resection including the safety zone was possible. With regard to
neck dissection, elective neck dissection is not required for normal
chondrosarcoma, but a definite pathological diagnosis was not obtained
at biopsy, and the prognosis is relatively poor.
Therefore, elective neck dissection was performed in this case be-
cause there was a possibility of mesenchymal chondrosarcoma and re-
constructive surgery was planned to reduce the dead space in the de-
fect. With regard to radiation therapy, it is currently used in positive
cases of stumps and in unresectable cases [6]. In the case of Ruark et al.
[36], Postoperative irradiation has been performed for positive stumps,
but all eight cases have died of underlying disease due to local recur-
rence, and Harwood et al. [38], Radiosensiti-vity was considered to be
low, as the annual survival rate was reported as 46 %. With regard to
chemotherapy, two types of typical systemic chemotherapy used for
soft tissue malignancies, CYVADIC therapy and MAID therapy, were
T. Inomata, et al. Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (xxxx) xxx–xxx

Fig. 5. Postoperative photograph of patient. Diffuse swelling was noted over outer cheek (arrow). (b) Postoperative six years by MRIT1-WI.

5
 T. Inomata, et al.
Table 1
Reported cases of chondrosarcoma of the condyler head.
First author, year Gender Age Clinical prezentation Imaging presentation Pathology Duration of symptoms Treatment Follow-up

1 Gingrass, 1966 [9] F 46 17 mmLMO, small swelling Condylar displaced posteriorly bone deposition S
2 Leiner, 1971 [10] F 48 2 cm mass, mild LMO Condylar resorbtion 24m S
3 Richter, 1974 [11] M 75 2 cm mass, diminished hearing TMJ space widening, opaque condyle, erosion of glenoid fossa, and increased 10m S 12m
condylar length
4 Nortje, 1976 [12] M 40 Slight facial asymmetry TMJ space widening, discrete radiopacities, and destruction of coronoid and well differentiated 6m S 24m
pterygoid plate
5 Morris,1987 [13] F 29 2.5 cm mass, no trismus Mass from condyle to infratemporal Low grade 24m S＋R 6m
6 Wasenko, 1990 [14] F 49 4 × 2.4 cm mass, conductive Mass from condyle to infratemporal fossa with calcification Low grade S
hearing loss
7 Nitzan, 1993 [15] F 36 7 mm LMO 4.5 cm hard mass Condyle displaced, resorbed condyle, external auditory canal, and middle Low grade 72m S
cranial fossa
8 Sesenna, 1997 [16] F 60 Painless mass, mild trismus Mass from condyle to infratemporal fossa with calcification GⅠ 12m S 7yr
9 Oda, 1998 [17] M 28 4 cm swelling Mass involving condyle, external auditory canal, base of skull, and bone 120m S
resorption
10 Batra, 1999 [18] M 65 2 cm mass, hearing loss Mass anterior to external ear canal encasing condyle and erosion of bone in well differentiated 18m S
middle cranial fossa
11 Mostafapour, 2000 [19] F 31 6 × 6 cm mass Left pterygoid space mass with involvment TMJ well differentiated 96m S
12 Mostafapour, 2000 [19] F 52 Mass with obstruction of external Mass on TMJ involving petrous temporal bone and middle fossa well differentiated 18m S＋R 6m
auditory canal
13 Yun, 2008 [20] F 29 20mmLMO, Palpable pain of Mass involving condyle with severe resorption GⅠ 120m S
preauricular area
14 Gallego, 2009 [21] M 54 Mild LMO with pain Mass involving condyle with severe reorption GⅠ 3m S 16m
15 Garzino-Demo, 2010 [22] F 65 Hard and painful mass, no trismus Mass centerd on TMJ with condylar resportion and calcification GⅠ 3m S＋R 9yr
16 González-Pérez, 2011 [23] M 57 Posterior open bite and crossbite Well-delimited lytic lesion involving condyle GⅠ 12m S 2yr
17 Xu, 2011 [24] M 34 3 mm LMO, hard, no tenderness or Large lobulated bone density mass in condyle and mandiblar ramus with GⅠ S
swelling calcification
18 Ramos-Murguialaday, 2012 M 45 No noteworthy fingings Osteolytic and expansive multilocular lesion encasing angle, ramus, and GⅡ Asymptomatic S 3yr
[25] condyle
19 Abu-Serriah, 2013 [26] M 48 Tenderness Mass from glenoid fossa with calcification, bone erosion of the middle cranial GⅡ 2m S 6m
fossa
20 Goutzanis, 2013 [3] M 23 Elastic, hard, no tenderness mass Mass involving condyle with bone destruction GⅠ-Ⅱ 2m S 2yr
21 MacIntosh, 2015 [27] F 31 5−8 mm LMO, tenderness Calcifying soft tissue mass medial to condyle Low grade 36m S 28yr
22 Kyu-Young, 2016 [28] F 60 LMO, 5.3 cm mass, swelling, pain, Lobulated mass with enhancing peripheral rim and innternal septa, condylar GⅠ 3yr S＋R 8m
parestesia resportion, adjacent bony erosion, periosteal reaction
23 Fukada, 2018 [29] F 78 4 × 3.6 cm mass, swelling Multilocular radioplaque mass involving condyle with resportion GⅡ 2m S 7yr
24 Slimani, 2019 [30] M 54 Swelling, Pain, laterodeviation Process centerd with calcifications, an osteolytic and osteocondensing aspect well differentiated 6yr S 10m
of the temporal bank and the condylar head
25 Present case, 2020 M 42 Swelling Mass involving irregular limbus and solid swelling with calcifications GⅠ 2m S 7yr

LMO:limited mouth opening, S:Surgery, R:Radiotherapy, m:months, yr:years.

6
T. Inomata, et al.
administered, but neither was tumor reduction, and Cisplatin and
Bleomycin alone were also administered. Although it has been reported
that the effect is not recognized, there is no report of effective che-
motherapy in chondrosarcoma. In our case, resection including sur-
rounding soft tissue was performed according to the malignant tumor
[39].
Postoperative open mouth and occlusal deviation were mild, and
neither eating nor swallowing disorder was observed, and only post-
opening training was performed to maintain QOL, and resection with a
histopathological safety margin Because it was completed, resection
and reconstruction seemed to be useful. Seven years after surgery, no
signs of recurrence or metastasis have been found. It is necessary to
carefully follow the presence or absence of local recurrence and distant
metastasis in the future.
Ethical approval
This study was approved by the Ethics Committee of International
University of Health and Welfare. The written approval was obtained
from the patient. (edited by Christina Gray, editorial office of
JOMSMP).
Declaration of Competing Interest
The authors have no conflicts of interest to declare.
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