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Table of Contents
CASE REPORT
Year : 2022  |  Volume : 24  |  Issue : 3  |  Page : 147-149

Chondroid tenosynovial giant cell tumor of the temporomandibular joint presenting as a deep-lobe parotid tumor


1 College of Medicine, Imam Mohammad Ibn Saud Islamic University, Riyadh, Saudi Arabia
2 Department of Otolaryngology-Head and Neck Surgery, College of Medicine, King Saud University, Riyadh, Saudi Arabia

Date of Submission22-Jun-2022
Date of Decision14-Jul-2022
Date of Acceptance19-Jul-2022
Date of Web Publication30-Sep-2022

Correspondence Address:
Dr. Reema Awad Alkhatabi
College of Medicine, Imam Mohammad Ibn Saud Islamic University, Riyadh
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sjoh.sjoh_27_22

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  Abstract 


Tenosynovial giant cell tumor (TGCT) is a rare benign craniofacial soft-tissue tumor characterized by overgrowth of the synovial tissue, tendon sheath, bursa, and joint structure. It is classified into two types, diffuse and localized TGCT, and has rarely been reported in the temporomandibular joint. The etiology of TGCT is unknown, and the management of TGCT varies depending on the tumor location and treatment-related morbidity. Here, we describe the case of a 47-year-old man who presented with a complaint of a right parotid mass that had slowly increased over 2 months and was associated with ear fullness. Initially, the lesion was painless, but pain during chewing without any limitation of the jaw movement developed subsequently. The patient was diagnosed with diffuse TGCT with chondroid metaplasia.

Keywords: Synovial membrane, temporomandibular joint, tenosynovial giant cell tumor


How to cite this article:
Alkhatabi RA, Aldhahri SF. Chondroid tenosynovial giant cell tumor of the temporomandibular joint presenting as a deep-lobe parotid tumor. Saudi J Otorhinolaryngol Head Neck Surg 2022;24:147-9

How to cite this URL:
Alkhatabi RA, Aldhahri SF. Chondroid tenosynovial giant cell tumor of the temporomandibular joint presenting as a deep-lobe parotid tumor. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2022 [cited 2022 Nov 28];24:147-9. Available from: https://www.sjohns.org/text.asp?2022/24/3/147/357637




  Introduction Top


Tenosynovial giant cell tumor (TGCT) is a rare benign craniofacial soft-tissue tumor characterized by overgrowth of the synovial tissue, tendon sheath, bursa, and joint structure.

The etiology of TGCT is unknown, but it could occur due to bleeding following trauma, lipid-metabolism disorders, inflammation, or neoplasms.[1],[2] The management of TGCT varies depending on the tumor location and treatment-related morbidity and ranges from simple surgical excision to extensive surgical resection with reconstruction. Radiotherapy or chemotherapy is used for recurrent disease or unresectable lesions. We present a case of TGCT-D suggest “diffuse TGCT” involving the right temporomandibular joint (TMJ) that presented as a parotid mass.


  Case Report Top


A 47-year-old otherwise healthy man presented with a complaint of a right parotid mass that had slowly increased over 2 months and was associated with ear fullness. Initially, the lesion was painless, but pain during chewing without any limitation of the jaw movement developed subsequently. The patient's past medical history and family history were unremarkable.

Clinical assessment revealed an approximately 3 cm × 3 cm, firm, nontender, deeply seated right parotid/preauricular mass with minimal mobility, intact facial nerve (VII) function, and no restriction of mandibular range of motion or lymphadenopathy.

Magnetic resonance imaging (MRI) revealed a right deep-parotid tumor. The tumor borders at the deep lobe of the parotid were unclear with high signal intensity in T2-weighted images, intermediate to low signal intensity on T1-weighted images, and some enhancement with gadolinium. The TMJ appeared normal, and no lymphadenopathy was noted [Figure 1]. Findings on computed tomography (CT) of the chest and abdomen were normal.
Figure 1: Magnetic resonance images (a), axial T2-weighted image (b and c) axial T1-weighted image, and (d) coronal T1-weighted image

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Fine-needle aspiration (FNA) showed atypical cells highly suggestive of a malignant salivary gland neoplasm.

The patient was scheduled for surgical resection including parotidectomy with selective neck dissection. Intraoperatively, the superficial lobe of the parotid was easily dissected off the VII nerve with no tumor seen. However, deep to the zygomaticofrontal branch of the VII nerve, the tumor was seen adherent to the deep lobe of the parotid, masseter muscle, and capsule of the TMJ. Complete resection was achieved by removing the gross tumor with the involved part of the masseter muscle and the TMJ capsule. Postoperatively, the patient recovered well and was discharged home on the next day. His mouth opening was normal with no TMJ symptoms. Weakness of the zygomaticofrontal branch completely resolved 4 weeks postoperatively.

Pathological examination showed a tumor measuring 3.5 cm in greatest dimension, and histopathological examination revealed a giant cell-rich lesion with irregular infiltration into the adjacent salivary gland, fibrofatty tissue with multiple reactive intraparotid lymph nodes, no lymphatic or vascular invasion, and no perineural invasion.

Immunohistochemical studies showed focal positivity for CD163 and D2-40 that confirmed the diagnosis of chondroid TGCT-D.

The patient's condition was discussed with the multidisciplinary tumor board team and the recommendation was to follow up the patient clinically and radiologically as needed with no need for adjuvant treatment. At 13 months postoperatively, the patient had no symptoms with normal physical examination, and follow-up CT and MRI showed a normal TMJ with no evidence of recurrent tumor.


  Discussion Top


The World Health Organization classifies giant cell tumors mainly based on whether the lesion originates from the tendon or the synovium.[3] It is classified based on the anatomical location (intra-articular or extra-articular) or growth pattern (localized or diffuse).[3] The localized form accounts for >75% of all TGCTs.[2] TGCT usually affects males and females equally in their third to fifth decades of life.[2] However, some studies reported that diffuse TGCT (TGCT-D) has a tendency to occur more frequently in younger individuals.

The difference between localized TGCT and TGCT-D is that TGCT-D is usually intra-articular with aggressive infiltrative behavior and a high recurrence rate.[1],[4] However, both types have the same microscopic features with overexpression of colony-stimulating factor.[1],[4]

The TMJ is a rare site of occurrence of TGCT, with <130 cases documented.[5] Although benign, TGCT is commonly aggressive, and in our patient, it invaded part of the masseter muscle and the TMJ capsule.

Clinically, TGCT usually presents as a nonspecific long-standing mass with or without pain may have some otological symptoms (hearing loss, ear fullness, and tinnitus), and may affect the facial nerve by direct compression.[6],[7] Due to the nonspecific symptoms, it can be misdiagnosed as other inflammatory or degenerative TMJ disorders.[8] Our patient belonged to the typical onset-age group. However, the symptoms and clinical and FNA findings were suggestive of a malignant deep-lobe parotid lesion.

On CT images, TGCT commonly appears as a soft-tissue mass with surrounding soft-tissue involvement and bone erosion with scalloped margins and occasional high-intensity areas due to hemosiderin deposition.[4] However, in our case, the mandible and TMJ were completely normal before and after the surgery on CT and magnetic resonance images.

The definitive diagnosis of TGCT is made based on the histopathological findings of the resected tumor, and because TGCT can resemble other cartilaginous diseases such as chondrosarcoma and synovial chondromatosis microscopically, the diagnosis is challenging.[5],[7] Therefore, radiologic findings, especially MRI findings are important for the interpretation of histopathologic findings. In our patient, despite the pathological findings and immunohistochemical positivity for CD163 and D2-40, we confirmed the diagnosis of TGCT after a thorough review of all radiological imaging studies by a head and neck and bone radiologist.

In all forms of TGCT, the treatment of choice is total surgical excision with free margins, and recently, good outcomes with regard to preventing postoperative recurrence have been reported with radiotherapy.[4],[8] Close follow-up with CT and MRI is recommended due to the high recurrence rate (approximately 20%–25%).[7] In this case, the treatment ended with total resection of the tumor, and no evidence of recurrence was noted at 13 months postoperatively.

In conclusion, we described a rare case of TGCT-D that presented as a parotid mass and was suspected as salivary gland neoplasm based on FNA biopsy findings. A careful review of radiological findings and a high index of suspicion can help clinicians and pathologists diagnose this rare tumor.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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 initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.



 
  References Top

1.
Yan H, Wang F, Xiang L, Zhu W, Liang C. Diffuse giant cell tumors of the tendon sheath in temporomandibular joint: Two case reports and review of the literature. Medicine (Baltimore) 2018;97:e11101.  Back to cited text no. 1
    
2.
Albakri A, Bakathir A, Al-Hashmi A, Al-Saadi N, Alshibani S, Al-kalbani F, et al. Chondroid tenosynovial giant cell tumor: A rare case involving the infratemporal fossa. Oman Med J 2021;36:e227.  Back to cited text no. 2
    
3.
Bredell M, Schucknecht B, Bode-Lesniewska B. Tenosynovial, diffuse type giant cell tumor of the temporomandibular joint, diagnosis and management of a rare tumor. J Clin Med Res 2015;7:262-6.  Back to cited text no. 3
    
4.
Shetty SK, Hegde U, Agarwal G, Sreeshyla HS. Chondroid tenosynovial giant cell tumor of temporomandibular joint. Ann Maxillofac Surg 2018;8:327-9.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Kanbour A, Hurrell M, Ricciardo P. Chondroid tenosynovial giant cell tumour of the temporomandibular joint. J Surg Case Rep 2021;2021:rjab155.  Back to cited text no. 5
    
6.
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. 6
    
7.
Yang X, Yao L, Yu T, Du X, Yue Q. Case report: Extra-articular diffuse tenosynovial giant cell tumor of the temporomandibular joint. Front Oncol 2021;11:643635.  Back to cited text no. 7
    
8.
Anbinder AL, Geraldo BM, Filho GR, Pereira DL, Almeida OP, Carvalho YR. Chondroid tenosynovial giant cell tumor of the temporomandibular joint: A rare case report. Braz Dent J 2017;28:647-52.  Back to cited text no. 8
    


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