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 Table of Contents  
CASE REPORT - TUMOR
Year : 2018  |  Volume : 8  |  Issue : 2  |  Page : 327-329

Chondroid tenosynovial giant cell tumor of temporomandibular joint


1 Department of Oral and Maxillofacial Surgery, JSS Dental College and Hospital, Mysuru, Karnataka, India
2 Department of Oral Pathology and Microbiology, JSS Dental College and Hospital, Mysuru, Karnataka, India

Date of Web Publication26-Dec-2018

Correspondence Address:
Usha Hegde
Department of Oral Pathology and Microbiology, JSS Dental College and Hospital, Mysuru, Karnataka
India
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DOI: 10.4103/ams.ams_161_18

PMID: 30693256

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  Abstract 


Tenosynovial giant cell tumor (TGCT), a benign proliferative lesion arising from the synovial membrane of the joints, is rarely seen in the temporomandibular joint. It frequently presents as a painful, preauricular swelling and affects the jaw functions. Two types of TGCT seen are diffuse TGCT (TGCT-D) and localized TGCT. A case of TGCT-D is described here, the highlight of the case being extensive areas of chondroid metaplasia which mimicked chondroid lesions, but was ruled out based on negative immunohistochemical findings within the tumor cells.

Keywords: Chondroid metaplasia, synovial membrane, temporomandibular joint, tenosynovial giant cell tumor


How to cite this article:
Shetty SK, Hegde U, Agarwal G, Sreeshyla H S. Chondroid tenosynovial giant cell tumor of temporomandibular joint. Ann Maxillofac Surg 2018;8:327-9

How to cite this URL:
Shetty SK, Hegde U, Agarwal G, Sreeshyla H S. Chondroid tenosynovial giant cell tumor of temporomandibular joint. Ann Maxillofac Surg [serial online] 2018 [cited 2019 Jun 26];8:327-9. Available from: http://www.amsjournal.com/text.asp?2018/8/2/327/248571




  Introduction Top


The pathological swellings in the temporomandibular joint (TMJ) can be due to various tumors and pseudotumors such as osteochondroma, chondrosarcoma, osteoid osteoma, plasmacytoma, synovial chondromatosis, eosinophilic granuloma, and pigmented villonodular synovitis.[1] Most of these tumors are rare and hence often clinically diagnosed as parotid mass or infection.[2] One example is tenosynovial giant cell tumor (TGCT), which is a rare benign proliferative growth of the synovium which shows destructive invasion by synovial-like mononuclear cells.[3]

TGCT, being one of the most common soft-tissue tumors of the hand (finger joints and tendon sheaths), is rarely seen in the head or neck region.[4],[5] According to the WHO classification of 2002 and 2013, the incidence rate in TGCT is not exactly known.[6] A total of 73 cases of TGCT affecting the TMJ have been reported in the English literature.[7] They are divided into four clinicopathological subtypes, according to their growth pattern (localized or diffuse) and anatomical site (intra- or extra-articular).[8] Difference between localized TGCT (TGCT-L) and diffuse TGCT (TGCT-D) is that TGCT-D has a tendency to occur in younger individuals, presents as a less well-defined soft-tissue mass, and has a locally aggressive growth and a high recurrence rate.[4],[5] This report presents a case of TGCT-D in the TMJ with chondroid metaplasia.


  Case Report Top


A 26-year-old male patient reported with the complaint of pain and swelling in the right pretragal region in 2015. The patient had a history of swelling and pain in the same region 5 years back, for which he had undergone curettage of the right condyle and superficial parotidectomy at a government medical institute. Histopathology report then showed normal salivary gland tissue and benign giant cell tumor of the condyle. The patient was asymptomatic until 4 months back when he noticed similar swelling associated with pain. On examination, a healed surgical scar was noted on the right preauricular region and extraoral diffuse swelling of about 4 cm in diameter in the right pretragal region [Figure 1]a and [Figure 1]b.
Figure 1: Frontal view of the clinical photograph showing extraoral diffuse swelling of about 4 cm in diameter in the right pretragal region (a). Lateral view of the lesion with healed surgical scar noted on the right preauricular region (b)

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Computed tomography (CT) showed a large expansile hypodense lytic lesion with lobulated margins measuring 2.7 cm × 2.9 cm × 2.6 cm involving the articular surface of the condylar process with thin bony septa within it [Figure 2].
Figure 2: Computed tomography scan showing a large expansile hypodense lytic lesion with lobulated margins measuring involving the articular surface of the condylar process

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Surgically, on inspection, the condyle was grossly deformed and reddish brown in color. Further anterior dissection was done till the coronoid process and associated part of superior ramus. Surgical resection of the right condyle and coronoid was done and reconstructed using costochondral rib graft [Figure 3]a and [Figure 3]b.
Figure 3: Intraoperative photograph showing grossly deformed condyle (a) and surgical resection of the right condyle and coronoid with reconstruction using costochondral rib graft (b)

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The histopathological examination of the biopsied tissue revealed a poorly circumscribed hypercellular connective tissue stroma [Figure 4]a. Stroma showed areas of hyalinization, consisting of lot of plump spindle-shaped cells and epitheloid mononuclear cells. It also showed extensive areas of hemosiderin pigmentation and lot of multinucleated giant cells [Figure 4]b. Diffuse areas of chondroid metaplasia were also evident within the stroma [Figure 4]c. Few hypocellular connective tissue areas were also evident [Figure 4]d. Based on the above findings, a diagnosis of chondroid TGCT was given. Immunohistochemistry (IHC) with CD68 was positive [Figure 5]a and negative with S-100 [Figure 5]b in the epitheloid mononucleated giant cells, further confirming the diagnosis of TGCT and ruling out chondroid lesions such as chondroblastoma and chondrosarcoma. The case has been followed up regularly, and the 2-year follow-up has been uneventful.
Figure 4: Sections reveal a poorly circumscribed hypercellular connective tissue stroma (a). Stroma showed areas of hyalinization, with lot of plump spindle-shaped cells, epitheloid mononuclear cells, extensive areas of hemosiderin pigmentation, and lot of multinucleated giant cells (b). Diffuse areas of chondroid metaplasia (c) and few hypocellular connective tissue areas were also evident (d) (H and E, ×100)

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Figure 5: The epitheloid cells show positive staining with CD68 (a) and negative with S-100 (b) (×100)

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  Discussion Top


TGCT belongs to a group of proliferative lesions affecting the synovial membrane and the tendon sheath, and it is rarely seen in the head or neck region. The etiology of TGCT is not certain. However, recent advances indicate that TGCT is a clonal neoplasm driven by overexpression of colony-stimulating factor-1.[2] TGCT-D differs in its clinical presentation from TGCT-L as it is more common in young people and presents as a poorly circumscribed mass with local aggressive biological behavior and high recurrence rates. Another distinct feature from TGCT-L is that it does not have a fibrous capsule.[4] The present case was a locally aggressive, recurrent growth in a young individual and did not show any fibrous capsule.

Radiologically, TGCT shows a soft-tissue mass often causing bone erosion and scalloped margins. CT scan frequently reveals cyst formation and areas of hyperattenuation due to hemosiderin deposition.[9] CT scan in our case revealed a large hypodense lytic lesion.

Histopathologically, TGCT shows features of synovial hyperplasia, with polyhedral synovial histiocyte-like cells, along with varying proportion of lymphocytes, giant cells, macrophages, and hemosiderin pigmentation.[3] A variant of TGCT called the “chondroid TGCT” in addition shows chondromyxoid, chondro-osseous, and hyaline-like matrix patterns.[10] The histopathology in the present case was suggestive of TGCT, with hyaline-like and chondroid areas. In view of the presence of large chondroid areas in the present case, IHC was done with CD68 and S-100. In TGCT, the histiocytes and multinucleated giant cells stain positively with CD68.[11] S-100 positivity in these cells is used to distinguish chondroid tumors which show positive staining.[12] Other markers that have shown varying positive expression are clusterin, desmin, CD163, CD68, and S-100. S-100-positive nuclei were found in the metaplastic chondrocytes in cartilaginous metaplasia, while the surrounding synovium-like monocytes, small histiocytoid cells, and multinucleated giant cells were S-100 negative.[7] In our case, the cells were CD68 positive and S-100 negative, thus ruling out the possibility of these cells being chondrocytic. Therefore, a final diagnosis of TGCT-D type with chondroid metaplasia was given based on the clinical, histopathological, and IHC findings.

Although surgical excision is considered as a preferred method of treatment for all forms of TGCT, it is influenced by the nature of the lesion. In most of the cases described in the literature, the treatment was with selective surgery, including enucleation of the neoplasm with wide surgical margins.[11] In the current case, since there was extensive recurrent involvement, resection of involved part was done and reconstructed to reduce the morbidity. During 2-year follow-up, there has been no evidence of recurrence.


  Conclusion Top


TGCT is a benign tumor which can be locally aggressive and a concern for high morbidity. Its occurrence in TMJ is rare, but establishing correct diagnosis aids in proper treatment planning and prognosis to the patient. In cases, where other features such as predominant chondroid areas cause difficulty in arriving at its definitive diagnosis, molecular investigations such as IHC with specific markers are advised.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Poveda-Roda R, Bagán JV, Sanchis JM, Margaix M. Pseudotumors and tumors of the temporomandibular joint. A review. Med Oral Patol Oral Cir Bucal 2013;18:e392-402.  Back to cited text no. 1
    
2.
De Benedittis M, Turco M, Petruzzi M, Cortelazzi R. Extra-articular diffuse-Type giant cell tumour of the temporomandibular joint. Int J Oral Maxillofac Surg 2013;42:380-5.  Back to cited text no. 2
    
3.
Fletcher CDM, Unni KK, Mertens F. Pathology and Genetics of Tumors of Soft Tissue and Bone. World Health Organization Classification of Tumors. Lyon: IARC Press; 2002. p. 110-4.  Back to cited text no. 3
    
4.
Martin RC 2nd, Osborne DL, Edwards MJ, Wrightson W, McMasters KM. Giant cell tumor of tendon sheath, tenosynovial giant cell tumor, and pigmented villonodular synovitis: Defining the presentation, surgical therapy and recurrence. Oncol Rep 2000;7:413-9.  Back to cited text no. 4
    
5.
Gong ZC, Lin ZQ, Moming A, Ling B, Liu H, Hu M, et al. Extra-articular diffuse tenosynovial giant cell tumour of the infratemporal fossa: Report of a case and literature review. Int J Oral Maxillofac Surg 2010;39:820-4.  Back to cited text no. 5
    
6.
Mastboom MJ, Verspoor FG, Verschoor AJ, Uittenbogaard D, Nemeth B, Mastboom WJ, et al. Higher incidence rates than previously known in tenosynovial giant cell tumors. Acta Orthop 2017;88:688-94.  Back to cited text no. 6
    
7.
Zhang C, Zhu L, Wang L, Hu Y, Li J, Tian Z. Diffuse tenosynovial giant cell tumor in the temporomandibular joint area in a Chinese population: A clinicopathological analysis of 32 cases. Int J Clin Exp Pathol 2016;9:1105-15.  Back to cited text no. 7
    
8.
Goldblum JR, Folpe AL, Weiss SW. Benign tumors and tumor-like lesions of synovial tissue. In: Goldblum JR, Folpe AL, Weiss SW, editors. Enzinger and Weiss's Soft Tissue Tumours. 6th ed. Philadelphia, PA: Saunders; 2013. p. 766-83.  Back to cited text no. 8
    
9.
Pina S, Fernandez M, Maya S, Garcia RA, Noor A, Pawha PS, et al. Recurrent temporal bone tenosynovial giant cell tumor with chondroid metaplasia: The use of imaging to assess recurrence. Neuroradiol J 2014;27:97-101.  Back to cited text no. 9
    
10.
Hoch BL, Garcia RA, Smalberger GJ. Chondroid tenosynovial giant cell tumor: A clinicopathological and immunohistochemical analysis of 5 new cases. Int J Surg Pathol 2011;19:180-7.  Back to cited text no. 10
    
11.
Tel A, Spinzia A, Boggio M. Diffuse tenosynovial giant cell tumour of the temporomandibular joint. Int J Oral Maxillofac Surg 2012;41:321-3.  Back to cited text no. 11
    
12.
Oda Y, Izumi T, Harimaya K, Segawa Y, Ishihara S, Komune S, et al. Pigmented villonodular synovitis with chondroid metaplasia, resembling chondroblastoma of the bone: A report of three cases. Mod Pathol 2007;20:545-51.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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