Pigmented villonodular synovitis of the temporomandibular joint: a case report and the literature review


Pigmented villonodular synovitis (PVNS) is an uncommon benign proliferative disorder of synovium that may involve joints, tendon sheaths, and bursae. It most often affects the knees, and less frequently involves other joints. It presents in the temporomandibular joints (TMJs) extremely rarely. The authors report an elderly female patient with PVNS of the TMJ with skull base extension, who had traumatic history in the same site. It was diagnosed through core-needle biopsy, which was not documented in the literature. Radical excision and follow-up for 7–8 years was recommended because of the reported malignant transformation and high recurrence rate. This case and previously reported cases in the literature are reviewed and discussed.

The term pigmented villonodular synovitis (PVNS) was introduced by J affe et al. in 1941 to describe a proliferative disorder of unknown aetiology involving diarthrodial joints, tendon sheaths and bursae . It’s an uncommon disorder, which has an annual incidence of 1.8 cases per million population . It most often affects the knees, including about 80% of cases . Other joints that may be affected by PVNS, in decreasing order of frequency, include the hips, ankles, small joints of the hands and feet, shoulders and elbows. Involvement of vertebral articular joints has also been documented . It presents extremely rarely in the temporomandibular joints (TMJs). The first reported occurrence was in 1973 by L apayowker et al. . To the authors’ knowledge, only 37 cases of PVNS affecting the TMJ have been reported in the English literature and 1 case in the Chinese literature; 34 of these were reported in detail ( Table 1 ). The authors present an additional case of PVNS affecting the TMJ, and review previous cases.

Table 1
Summary of reported cases of PVNS involving the TMJ.
A. Clinical features and preoperative diagnosis
No. Authors/date Age (y);sex;site Clinical features Duration Bone destruction Preoperative diagnosis
1 L apayowker et al. 1973 22;M;L PS, masticatory pain 18m TMJ Malignant PT
2 L apayowker et al. 1973
D inerman and M yers 1977
58;F;L Clicking; tinnitus; hearing diminution 1y TMJ, ramus, SBICE, EAC Biopsy: fibroadipose tissue
Open biopsy: PVNS
3 B arnard 1975 37;M;L PS, pain, LMO, MD to L 3w TMJ Infection arthritis or neoplasia
4 M iyamoto et al. 1977 34;M;R PS 1.5y TMJ PT
5 T akagi and I shikawa 1981 36;M;R Oppressive pain, clicking, LMO 3y TMJ NR
6 R ickert and S hapiro 1982 39;F;L Parotid mass 1m TMJ PT; FNA: giant cell granuloma, ‘brown tumour’, neoplasm
7 G allia et al. 1982
C urtin et al. 1983
47;F;L PS, LMO, masticatory pain, MD to L, otalgia 2y TMJ PVNS
8 O’S ullivan et al. 1984
B ertoni et al. 1997
61;F;L Swelling in parotid area NR TMJ, coronoid process, ramus, zygomatic arch, EAC, SBICE Benigh PT
9 D awiskiba et al. 1989 32;M;R TMJ swelling, pain, LMO NR No FNA: PVNS
10 E isig et al. 1992 50;F;R HL, mass in ear canal 2m TMJ, SBICE, EAC, mastoid, middle ear ossicles Central giant cell granuloma
11 S yed et al. 1993 10;F;L PS 1y No PT
12 F ranchi et al. 1994 59;F;L Swelling in parotid area, pain 6m No PT.
13 Y oussef et al. 1994 41;F;L TMJ swelling, pain, LMO, decreased lateral excursion to L 6m No Incisional biopsy: PVNS
14 T anaka et al. 1997 47;M;R PS, masticatory pain, LMO, MD to R 8m TMJ Giant cell-containing lesion of TMJ
15 R enaga et al. 1997 70;F;L TMJ swelling, intermittent pain, LMO, clicking 7m TMJ PVNS
16 Y u et al. 1997 48;M;R PS, pain when wide mouth opening 7m TMJ PT
17 C how et al. 1998 42;F;R Zygomatic mass 6m TMJ Giant cell tumour, chondroblastoma
18 S ong et al. 1999 57;F;R TMJ swelling 5y TMJ PT
19 B emporad et al. 1999
S tryjakowska et al. 2005
37;M;R PS, pain, trismus, tinnitus, HL 4y TMJ, SBICE, maxilla, maxillary sinus, carotid canal, EAC PT; Incisional biopsy: ‘brown tumour’; FNA: PVNS
20 L ee et al. 2000 59;F;R PS, decreased salivary flow 6y TMJ PT; Punch biopsy: PVNS
21 K isnisci et al. 2001 45;F;L Cheek swelling, pain, LMO, MD to L NR No NR
22 S hapiro et al. 2002 36;M;R Temporal mass, clicking, some HL, popping sensation in ear NR SBICE Malignant process; CT guided FNA: benign giant cell-containing lesion
23 C hurch et al. 2003 42;M;R PS, clicking, HL, mass nearly occluded EAC 4m TMJ, zygomatic arch, SBICE, EAC Biopsy: PVNS
24 C hurch et al. 2003 33;M;R PS, deep pain, trismus 2y TMJ FNA: PVNS
25 T osun et al. 2004 60;M;L TMJ mass, V2/V3 numbness/paresthesia, LMO, MD to L, HL, otitis media 10y TMJ, ramus, SBICE, carotid canal, foramen ovale, EAC, sphenoid bone Otitis, PT; FNA: PVNS
26 A oyama et al. 2004 33;M;R PS, clicking > 20y, LMO NR TMJ, skull base Malignant tumour, giant cell tumour; Biopsy: PVNS
27 C ascone et al. 2005 38;F;L PS, TMJ pain NR No FNA: non-specific arthritis
28 S tryjakowska et al. 2005 36;F;R Painful parotid lesion, radiating pain 4m No PT
29 F ang et al. 2007 44;M;R PS 2m TMJ, zygomatic arch Pleomorphic adenoma; FNA: PT
30 C ascone et al. 2008 78;M;R PS, orofacial pain, LMO, MD to R NR No NR
31 D ay et al. 2008 38;M;L Mass in zygomatic process area, pain 1m SBICE, foramen ovale, vidian canal, foramen rotundum Cholesterol cyst, vascular bone tumour
32 S hkoukani et al. 2009 74;F;R PS, pain, LMO Few m TMJ CT-guided FNA: PVNS
33 C ai et al. 2009 21;F;L TMJ pain, LMO, clicking, MD 3m No Anterior disc displacement without reduction
34 H erman et al. 2009 36;M;L PS, pain, numbness, LMO, MD to L, otalgia 12–18m Zygomatic arch, glenoid fossa, SBICE Biopsy: PVNS

B. Treatment and prognosis
No. Treatment Complications and prognosis Rec. (FU-period) [Rec. time]
1 CE Uneventful No
2 CE, Condy., partial Parot., craniectomy, reconstructed with free graft of fascia temporalis NR No (4y)
3 Incisional biopsy Some persistent MD to L NR (6m)
4 CE Temporary tinnitus, EAC narrowing No (2y)
5 1st–2nd: CE, Condy. Uneventful Yes [5y]; no Rec. (11y)
6 Amputated tumour from TMJ, superficial Parot. Temporary FP, limited jaw motion No (3m)
7 CE Temporary FP, occlusal discrepancy No (2y)
8 1st–3rd: mass excision, Parot., Mand., craniectomy; 4st: RT; 5th: surgery Dead 20d after last operation with malignant transformation, intracranial extension and lung metastasis Yes [1.5y, 2.5y, 2y, 4y]
9 Surgery (not specified) NR NR
10 CE, craniectomy, Condy., radical mastoidectomy, repaired dura with temporal fascia graft Minimal MD No (1y)
11 CE, superficial Parot. Temporary FP NR (8w)
12 CE NR No (1y)
13 CE, superficial Parot. Uneventful No (14m)
14 CE, craniectomy, Condy. Temporary FP No (2y)
15 CE Uneventful No (3y)
16 1st: superficial Parot.; 2st: surgical exploration NR NR
17 CE, Condy. Uneventful No (2y)
18 CE, total Parot., Condy. NR NR
19 Embolization, intradural resection, Mand., total Parot., facial nerve grafting, suprahyoid neck dissection, repaired with pericranial graft and rectus myocutaneous free pedicle flap FP No (3y)
20 Remove mass and involved facial nerve, sural nerve grafting, Condy., reconstructed with costochondral graft Minimal MD; FP No (2y)
21 CE Uneventful No (1y)
22 Embolizaiton, CE, craniotomy Uneventful No (7y)
23 Wide excision, partial Mand, craniectomy Uneventful No (3y)
24 Wide excision Uneventful No (2y)
25 CE, Condy., temporal craniotomy, partial Parot. MD to R No (6y)
26 CE, Condy., craniotomy, covered dura by temporal muscle pedicle flap Preauricular concavity; temporary FP; MD to R No (2y)
27 CE, superficial Parot. Uneventful No (1y)
28 1st–2nd: Parot., CE Uneventful Yes [15m]; no Rec. (1y)
29 CE NR No (20m)
30 CE Uneventful No (2y)
31 CE, repaired temporal bone defect and tegmen tympani with titanium mesh Temporary HL NR (3m)
32 CE, tracheostomy tube placement, superficial Parot., Condy., partial Mand. Temporary FP; MD No
33 Removed lesions with arthroscopy Uneventful No (13m)
34 CE, craniectomy; repaired with cranial bone, lateral pterygoid and temporalis muscles Uneventful No (11y)
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Jan 27, 2018 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Pigmented villonodular synovitis of the temporomandibular joint: a case report and the literature review
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