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BUI ET AL 405

rome) due to the raised resistance to vitamin D acquired aboutthe age of 15 years. Q J Med 16:33, 1947

4. Prader A, Illig R, Uehlinger RE, et al: Rickets caused by bonetumors. Helv Pediatr Acta 14:554, 1959

5. Cai Q, Hodgson SF, Kao PG, et al: Inhibition of renal phosphatetransport by a tumor production in a patient with oncogenicosteomalacia. N Engl J Med 330:1645, 1994

6. Ogose A, Hotta T, Emura I, et al: Recurrent malignant variant ofphosphaturic mesenchymal tumor with oncogenic osteomala-

cia. Skeletal Radiol 30:99, 2001

Kamal F. Busaidy, BDS, FDSRCS,

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awawoi:10.1016/j.joms.2008.01.043

7. Weidner N, Santa Cruz D: Phosphaturic mesenchymal tumors:A polymorphous group causing osteomalacia or rickets. Cancer59:1442, 1987

8. Weidner N: Review and update: Oncogenic osteomalacia-rick-ets. Ultrastruct Pathol 15:317, 1991

9. Siegel H, Rock MG, Inwards C, et al: Phosphaturic mesenchy-mal tumor. Orthopedics 25:1279, 2002

0. Ungari C, Rocchi G, Rinna C, et al: Hypophosphaturic mesen-chymal tumor of the ethmoid sinus associated with oncogenic

osteomalacia. J Craniofac Surg 15:523, 2004

J Oral Maxillofac Surg67:405-409, 2009

Chondroblastoma of theTemporomandibular Joint: Report of a

Case and Literature ReviewPeter Bui, DDS, MD,* Doina Ivan, MD,† Dwight Oliver, MD,‡

§ and James Wilson, DDS¶

hondroblastoma is a rare benign tumor of the headnd neck. We present a case of chondroblastoma ofhe temporomandibular joint (TMJ) with literatureeview and analysis of reported cases involving theMJ. Our case is unique because the tumor is ex-

raosseous in nature. It arises from either the menis-us or posterior ligament. Although resection haseen the recommended treatment as documented inrthopedic and otolaryngology literature, conserva-ive treatment such as excision and curettage to pre-erve critical anatomical structures has been carried

*Chief Resident, Department of Oral & Maxillofacial Surgery, The

niversity of Texas Health Science Center at Houston Dental

ranch, Houston, TX.

†Assistant Professor, Department of Pathology & Lab Medicine,

he University of Texas M.D. Anderson Cancer Center, Houston,

X.

‡Assistant Professor, Department of Pathology & Lab Medicine,

he University of Texas Health Science Center at Houston Medical

chool, Houston, TX.

§Assistant Professor, Department of Oral & Maxillofacial Surgery,

he University of Texas Health Science Center at Houston Dental

ranch, Houston, TX.

¶Associate Professor and Vice-Chair, The University of Texas

ealth Science Center at Houston Dental Branch, Houston, TX.

Address correspondence and reprint requests to Dr Bui: UT-HSC

t Houston Dental Branch, Oral and Maxillofacial Surgery, 6516 MD

nderson Blvd, Ste 2.059, Houston, TX 77030; e-mail: peter.q.bui@

th.tmc.edu

2009 American Association of Oral and Maxillofacial Surgeons

278-2391/09/6702-0025$36.00/0

ut successfully. Frequent clinical and radiographicollow-ups are essential for early detection and man-gement of recurrence.

Chondroblastoma was first described by Jaffe andichtenstein in 1942.1 It is a rare, benign, cartilagi-ous tumor, occurring infrequently in the head andeck area. It comprises about 1% of all benign boneumors and is found usually in the epiphysis of longones such as the distal femur, proximal tibia, androximal humerus.2 There has been only 1 reportedase of chondroblastoma of the TMJ that was ex-raosseous in nature.3 The following report is anotherase of extraosseous chondroblastoma of the TMJ thatas treated successfully with excision and curettage.

eport of a Case

A 53-year-old woman was referred to the Oral and Max-llofacial Surgery department at the University of Texasealth Science Center in Houston from an otolaryngologist

or evaluation of a right TMJ mass that was detected withomputed tomography (CT) and magnetic resonance imag-ng (MRI). The patient had a 1-year history of intermittentebilitating pain in the right pre-auricular area, accompa-ied by episodic trismus and subjective swelling in the area.he symptoms had worsened progressively during the pastmonths. She denied any history of facial trauma, head-

che, facial paralysis, paresthesia, dysphagia, drainage oremorrhage from the ears or nose, hearing loss, locking orrepitus of the TMJ, or malocclusion.Her past medical history was significant for hypertension

nd non-insulin dependent diabetes mellitus (NIDDM) forhich she took verapamil, hydrochlorothiazide, glipizide,

nd metformin. She had a hysterectomy 11 years previously

ithout complication.

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406 CHONDROBLASTOMA OF THE TMJ

Physical examination of the patient showed normal ap-earance of the external auditory canals and tympanicembranes. Masticatory muscles were not tender to palpa-

ion. Occlusion was stable and repeatable. Maximal inter-ncisal opening was 55 mm. There was no tenderness oreviation on mouth opening or closing. Maximal excursiveovements were 8 mm to the left and 8 mm to the right.aximal protrusive movement was 2 mm. The maxillary

hird molars, the mandibular left third molar, and the man-ibular right second molar were missing. The remainingeeth were in a good condition. There was no palpable headr neck mass, or lymph node enlargement. Examination ofhe cranial nerves showed no abnormality.

Panorex, MRI, and CT scans of the TMJ showed a softissue mass in the right glenoid fossa eroding the posteriorurface of the fossa. There was a profound thinning of theone without protrusion of the mass into the middle cranialossa (Figs 1-3).

The differential diagnosis included chondrosarcoma,hondromyxoid fibroma, synovial sarcoma, synovial chon-romatosis, and aneurysmal bone cyst.4

The patient was taken to the operating room wherexcision of the lesion was carried out via a pre-auricularpproach. The lesion extended from the retrodiscal tissueo posterior glenoid fossa and into the osseous portion ofhe squamotympanic fissure. Excision of the tumor, curet-age of the fossa, and partial meniscotomy were carried out.ultiple specimens from the remaining meniscus, posterior

igament, and bone were submitted to the pathology lab forrozen sections. The biopsy specimens showed margins thatere free of tumor.Grossly, the definitive pathology specimens showed that

he tumor involved the posterior ligament and the meniscusf the TMJ. The chondroblastoma was focally attached to

ntact bone and multiple sections of the specimen failed tohow tumor within the bone. Histologically, the lesiononsisted of fibroconnective tissue with different sized is-ands of proliferating chondroblasts in an eosinophilic car-ilaginous matrix (Fig 4). The neoplastic cells within theslands had sharply defined cell borders and eosinophilic,ocally clear cytoplasm. The nuclei were mildly pleomor-hic, ovoid to round, variably sized, and frequently hadlefts or small folds and a small nucleolus. The chromatin

IGURE 1. Portion of Panorex showing a lesion in the right glenoidossa.

ui et al. Chondroblastoma of the TMJ. J Oral Maxillofac Surg009.

B2

attern was vesicular to slightly granular. Only rare mitoticgures were observed. Multinucleated, osteoclast-like cellsere scattered among the neoplastic cells, and calciumeposits around degenerating individual cells produced theharacteristic “chicken-wire” appearance of chondroblas-oma (Fig 5). No areas of osteoid formation or necrosis weredentified. An immunohistochemical stain for S-100 protein

as positive on the neoplastic cells within and outside theartilaginous matrix, showing their chondroid differentia-ion.

The patient healed uneventfully and is free of tumor at 4ears.

iterature Review

There have been 12 cases of chondroblastoma ofhe TMJ reported in English literature.3,5-14 Several of

IGURE 2. Axial view of CT showing bony resorption of the rightlenoid fossa.

ui et al. Chondroblastoma of the TMJ. J Oral Maxillofac Surg009.

IGURE 3. Coronal view of MRI with gadodiamide contrast show-ng bony resorption of the right glenoid fossa.

ui et al. Chondroblastoma of the TMJ. J Oral Maxillofac Surg009.

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BUI ET AL 407

hese reports did not include complete descriptionsf symptomatology and modes of treatment.5,7

AGE AND GENDER DISTRIBUTION

The patients ranged from 27 years to 56 years ofge with a mean age of 39. The ratio of male to femaleas 1:1.4.

PRESENTING SYMPTOMS

Common presenting symptoms were swelling,ain, and decreased range of motion. The duration ofhese symptoms ranged from 1 to 3 years beforereatment. On average, patients had 3 symptoms (Ta-le 1).3,5,7-9,11-14

IGURE 4. Chondroblastoma. Islands of proliferating mononu-lear and multinucleated neoplastic cells in an eosinophilic carti-aginous matrix.

ui et al. Chondroblastoma of the TMJ. J Oral Maxillofac Surg009.

IGURE 5. Chondroblastoma. Characteristic “chicken-wire” cal-ification surrounds individual tumor cells.

ui et al. Chondroblastoma of the TMJ. J Oral Maxillofac Surg009.

B2

PHYSICAL FINDINGS

Swelling was the predominant finding. Cases re-orted by Bertoni et al and Dahlin and Ivins were not

ncluded here because the physical findings were notocumented (Table 2).3,5,7-9,11-14

RADIOGRAPHIC FINDINGS

Recent reports used more than 1 imaging modalitynd their descriptions were more detailed. CT scansontributed to two thirds of imaging modalities se-ected. Only 2 studies used MRI to evaluate the extentf the lesion (Table 3).3,5,7-9,11-14

MANAGEMENT

Reported treatments included curettage, resection,nd excision. In some reports, the forms of treatmentere not well described. There was a recurrence after

he first curettage in 1 report but there was no furtherecurrence at 3.5 years after a second curettage. Nonef the cases treated by resection recurred althoughhere was a lack of follow-up found in 1 report (Ta-le 4).3,5,7-9,11-14

iscussion

Since first being described by Jaffe and Lichtensteinn 1942, about 58 chondroblastoma cases have beenocumented from the craniomaxillofacial region,ith 12 cases involving the TMJ area.2,3,5,6,8,9,11-16 Tour knowledge, only 1 other case published in English

iterature has been found to have extraosseous origin

Table 1. PRESENTING SYMPTOMS OF THE PATIENTS

Symptoms Patients (n � 12)

welling 9ain 8rismus 5licking 2earing loss 2talgia 1aresthesia 1

ui et al. Chondroblastoma of the TMJ. J Oral Maxillofac Surg009.

Table 2. PRESENTING FINDINGS OF THE PATIENTS

Findings Patients (n � 7)

welling 5rismus 4eviation of mandible 3alocclusion 1repitation 1

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408 CHONDROBLASTOMA OF THE TMJ

ssociated with the TMJ. This lesion was found in theMJ ligament of a 31-year-old woman with a 2-yearistory of recurrent left facial swelling and pain. Itas removed via en bloc resection without recur-

ence at a 15-month follow-up visit.3 Our case isnique because the tumor had invaginated intactone without any evidence of infiltration and it wasreated successfully with a more conservative ap-roach involving excision and curettage.Our case report is a third case that used MRI as a

iagnostic tool, which shows superb images of soft

Table 3. RADIOGRAPHIC DESCRIPTION OF THE LESION

AuthorPanorex/Plain

Films CT MRI

oodsell and Hubinger9 X Expdo

ahlin and Ivins7 X Tumpahr et al3 X Abu

mertoni et al5 (4 cases) X Noayne and Yusuf13 X Des

boongo et al12 X Smaoga et al8 X X CT:

MRIan

ondoh et al11 X X Pande

CT:m

audet et al14 X X X Panjo

CT:rigr

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ui et al. Chondroblastoma of the TMJ. J Oral Maxillofac Surg 2

Table 4. TREATMENT MODALITIES AND THEIR OUTCOM

Author Treatmen

oodsell and Hubinger9 First curettageSecond curettage

ahling and Ivins7 No documentation of trpahr et al3 En bloc resection of maertoni et al5(4 cases) Case 1: Wide excision

Case 2: No documentatiCase 3: CurettageCase 4: Marginal excisio

oga et al8 Resectionongo et al12 Subtotal excision of conayne and Yusuf13 Resectionondoh et al11 Resectionaudet et al14 Resection

ui et al. Chondroblastoma of the TMJ. J Oral Maxillofac Surg 2009.

issues that is necessary to identify and delineate theumor before surgery. It also has other advantagesuch as the avoidance of ionizing radiation, fewerrtifacts from metal dental filling, crowns, or prosthe-is.

Tumors of the TMJ are rare, and chondrogenicumors in this location are extremely uncommon.ecause chondroblastoma has been reported so infre-uently in the TMJ, more common entities should firste considered in the differential diagnosis of masses

n this location. Osteochondroma is the most frequent

Description

mass from the condylar neck to the sigmoid notchthe ramus

panded the condyleof tumor against the zygoma, pterygoid plate, and

cranial fossad description was givenn of posterior aspect of the right condyle with an area oflerosis anterior to itirregularities of the condyle

cence of the tympanic membranensile mass in the infratemporal region invading the TMJial baselytic defect in posterior portion of the condyle, well

ted radiolucencyntinuity of the cortex of posterior condyle; expandingposteromedial directionnterior condylar destruction with a scalloping effect; the

ace was enlargedction of the squamous portion of the temporal bone, thenoid fossa, the right zygomatic arch, and the right

wing of the sphenoidsement of the right mandibular condyle by the lateralf a mass that has destroyed the right glenoid fossa

nce imaging.

Outcome

Recurrence at 3 yearsNo recurrence at 3.5 years

tNo recurrence at 15 months follow-upNo recurrence at 13 years

No documentation of follow-upNo recurrence at 17 monthsNo recurrence after 4 years

nd fossa No recurrence after 12 monthsNo dataNo recurrence after 12 yearsNo recurrence after 4 years

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BUI ET AL 409

one neoplasm in the TMJ. It contains trabecularone with a rim of cartilage but lacks the chondro-last proliferation and chondroid matrix characteris-ic of chondroblastoma. Radiopaque structures areommonly identified on CT scan. Chondrosarcoma,rises in the head and neck region in about 1% ofases. This malignant tumor is invasive, may metasta-ize, and contains malignant-appearing chondrocytes.artilaginous matrix within abundant fibrous tissueuggests a diagnosis of chondromyxoid fibroma, aistogenetic relative of chondroblastoma that isqually rare in the TMJ. However, paucity of myxoidreas and abundant individual cell calcifications areot features of chondromyxoid fibroma.4 Synovial sar-oma, a tumor derived from pluripotent mesenchymeells capable of periarticular tissue differentiation, islso rare in the TMJ. In the majority of cases, theresence of a biphasic pattern (spindle cells and ep-

thelioid cells) is described. Furthermore, they do notxhibit chondroid differentiation. More common inhe TMJ region are non-neoplastic processes such asynovial chondromatosis, ganglion cyst, and aneurys-al bone cyst, which should also be considered in the

ifferential diagnosis. Cartilaginous nodules in theMJ space, representing metaplasia of mesenchymalells, characterize synovial chondromatosis. Thehondrometaplasia nodules, which can become looseodies within the joint space, are covered by synovialells and can undergo mineralization of the matrixnd exhibit nuclear atypia. However, no chondroblastell proliferation is present.4 A lack of cystic cavitiesnd blood-filled spaces excludes synovial, ganglion,nd aneurysmal bone cysts from the diagnosis.

Due to the rarity of chondroblastoma in the TMJrea and a lack of long-term follow-up in many re-orted cases, it is difficult to assess the success ofifferent treatment modalities. Unlike giant cell tu-ors, the chondroblastoma is non-aggressive and can

e cured by conservative treatment. Dahlin et al,8 in atudy of 125 cases of chondroblastoma in long bones,oncluded that curettage effects cure in about 90% ofases. Curettage is recommended when infiltration ofone is limited or has not occurred.7 Choosing toesect the tumor in our case would have involved araniectomy resulting in more prolonged recoverynd increased morbidity. A follow-up regimen includ-

ng CT imaging at 6 months and annually for 5 years

as been recommended, as recurrence seems to oc-ur within this time range. Radiation therapy has beenuggested as an alternative treatment in cases whereurgery might be associated with significant morbid-ty, for example due to location of the lesion, or as andjunctive therapy to curettage of chondroblastoma.owever, the risk of malignant transformation with

adiation therapy, although very low, must be consid-red. The incidence of post-irradiation sarcomas isstimated to be between 0.03% to 0.8%.6-8,17

eferences1. Jaffe HL, Lichtenstein L: Benign chondroblastoma of bone. A

reinterpretation of the so-called calcifying or chondromatousgiant cell tumor. Am J Pathol 18:969, 1942

2. Varvares MA, Cheney ML, Goodman ML, et al: Chondroblas-toma of the temporal bone. Case report and literature review.Ann Otol Rhinol Laryngol 101:763, 1992

3. Spahr J, Elzay RP, Kay S, et al: Chondroblastoma of the tem-poromandibular joint arising from articular cartilage: A previ-ously unreported presentation of an uncommon neoplasm.Oral Surg 54:430, 1982

4. Warner BF, Luna MA, Newland JR: Temporomandibular jointneoplasms and pseudotumors. Adv Anat Pathol 7:365, 2000

5. Bertoni F, Unni KK, Beabout JW, et al: Chondroblastoma of theskull and facial bones. Am J Clin Pathol 88:1, 1987

6. Blaauw G, Prick JJ, Versteege C: Chondroblastoma of the tem-poral bone. Neurosurgery 22:1102, 1988

7. Dahlin DC, Ivins JC: Benign chondroblastoma: A study of 125cases. Cancer 30:401, 1972

8. Goga D, Fassio E, Fetissof F, et al: Chondroblastoma of thetemporomandibular region. J Oral Maxillofacial Surg 57:1270,1999

9. Goodsell JO, Hubinger HL: Benign chondroblastoma of man-dibular condyle: Report of a case. J Oral Surg 22:355, 1964

0. Kingsley TC, Markel SF: Extraskeletal chondroblastoma: A re-port of the first reported case. Cancer 27:203, 1971

1. Kondoh T, Hamada Y, Kamei K, et al: Chondroblastoma of themandibular condyle: Report of a case. J Oral Maxillofacial Surg60:198, 2002

2. Longo F, Califano L, Zupi A, et al. Chondroblastoma of thetemporomandibular joint: Case report with cytopathologic andhistopathologic study. J Oral Maxillofac Surg 57:1372, 1999

3. Payne M, Yusuf H: Benign chondroblastoma involving the man-dibular condyle. Br J Oral Maxillofacial Surg 25:250, 1987

4. Gaudet EL, Nuss DW, Johnson DH, et al: Chondroblastoma ofthe temporal bone involving the temporomandibular joint,mandibular condyle, and middle cranial fossa: Case report andreview of the literature. J Craniomandibular Pract 22:160, 2004

5. Cohen B, Kan FH, Dardy DJ: Arthroscopic resection of chon-droblastoma of the knee. Arthroscopy 8:370, 1992

6. Coleman SS: Benign chondroblastoma with recurrent soft tis-sue and intraarticular lesions: Report of a case. J Bone JointSurg 48A:1554, 1966

7. Mark RJ, Poen J, Tran LM, et al: Post-irradiation sarcomas. A

single institution study and review of the literature. Cancer73:2653, 1994