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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