Abstract
Background
Pigmented villonodular synovitis (PVNS) is a rare/benign condition of the synovial joint lining. It most commonly presents in the knee but has also been reported to occur in the temporomandibular joint (TMJ). Although there are several series reporting the use of postoperative radiotherapy (PORT) for extremity PVNS, there is scant literature on the use of PORT for PVNS of the TMJ.
Methods
We conducted a literature review for case reports related to PVNS of the TMJ and discuss two additional cases treated with surgery and PORT.
Results
71 cases were found in the literature. 89% were the diffuse subtype. 92% had primary surgery and 7% had PORT. 68% were locally controlled. Both patients treated at our institution are locally controlled.
Conclusions
PVNS of the TMJ is a rare entity. Surgery is the mainstay of treatment but PORT may be useful for local control of extensive tumors or positive margins.
1
Introduction
Pigmented villonodular synovitis (PVNS) is a benign, but locally aggressive, overgrowth and inflammation of the synovium that results in invasion and destruction of adjacent bone and functional impairment . The disease is rare with an annual incidence of 1.8 cases per million population . PVNS is almost always monoarticular, with 80% of cases occurring in the knee . Involvement by PVNS of the temporomandibular joint (TMJ) is uncommon, with the first case reported by Lapayowker et al. in 1973 . There are two forms of PVNS: localized (nodular) and diffuse. The diffuse subtype is more common and involves the entire synovium. Both forms of the disease are primarily treated with surgery – a marginal resection for the nodular subtype and a total or near total synovectomy for the diffuse subtype . The local recurrence rate is low for the nodular subtype but higher in the diffuse subtype (up to 45%). Because of the high risk of recurrence of the latter type, postoperative radiotherapy has been advocated and has been shown to improve local control in several small retrospective series of patients with diffuse PVNS of the extremities . In this report we conducted a literature review, present two additional cases, and discuss the role of radiation therapy in the management of diffuse PVNS of the TMJ.
2
Materials and methods
A PubMed query was performed using the keyword combinations of pigmented villonodular synovitis temporomandibular joint and the synonymous term of tenosynovial giant cell tumor temporomandibular joint . Articles in English and non-English (translated) reporting on at least one case of PVNS of the TMJ were included. The references and literature review tables of these articles were also reviewed to ensure all pertinent literature was included. A total of 71 cases were identified in the literature, not including the two cases presented in this report. The details are represented in Table 1 .
No. | Author (date) | Age (y)/ gender | Type of PVNS | Surgery | Radiation (Gy) | Local recurrence/follow-up period | Other notes |
---|---|---|---|---|---|---|---|
1–2 | Lapayowker et al. (1973) | 22/M | Diffuse | Yes | No | No/8 y | |
58/F | Diffuse | Yes | No | NR | |||
3 | Barnard (1975) | 37/M | NR | Conservative approach, biopsy only | No | No change in lesion/6 mo | |
4 | Dinerman and Myers (1977) | 58/F | Diffuse | Yes | No | No/4 y | |
5 | Miyamoto et al. (1977) | 34/M | Diffuse | Yes | No | No/2 y | |
6 | Raibley (1977) | 62/F | Localized | Yes | No | No/5 mo | With synovial chondromatosis |
7 | Makek and Drommer (1978) | 55/F | Localized | Yes | No | No/9 mo | |
8 | Geiger and Pesch (1980) | 50/F | Diffuse | Yes | No | No/4 y | |
9 | Takagi and Ishikawa (1981) | 36/M | Diffuse | Yes, X2 | No | Yes/5 y; No/11 y | |
10 | Gallia et al. (1982) | 47/F | Diffuse | Yes | No | No/2 y | |
11 | Rickert and Shapiro (1982) | 39/F | Diffuse | Yes | No | NR | |
12 | Curtin et al. (1983) | 47/F | Diffuse | Yes | No | NR | |
13 | O’Sullivan et al. (1984) | 61/F | Diffuse | Yes, X3 | Yes, after the fourth recurrence (40) | Yes/X3 in a 7 y period | |
14 | Dawiskiba et al. (1988) | 32/M | Diffuse | NR | No | NR | |
15 | Eisig et al. (1992) | 50/F | Diffuse | Yes | No | No/1 y | |
16 | Syed et al. (1993) | 10/F | Diffuse | Yes | No | NR | |
17 | Franchi et al. (1994) | 59/F | Diffuse | Yes | No | No/1 y | |
18 | Shapiro et al. (1996) | 36/M | Diffuse | Yes | No | No/18 mo | |
19 | Youssef et al. (1996) | 41/F | Localized | Yes | No | No/14 mo | |
20 | Allias-Montmayeur et al. (1997) | 39/F | Localized | Yes | No | No/7 mo | |
21 | Bertoni et al. (1997) | 71/F | Diffuse, metastatic | Yes, x5 | Yes (45) | Yes/X4 in 7 y period | Malignant PVNS, local disease, intracranial extension, and lung metastasis |
22 | Renaga Rubin et al. (1997) | 70/F | Diffuse | Yes | No | No/3 y | |
23 | Tanaka et al. (1997) | 47/M | Diffuse | Yes | No | No/2 y | |
24 | Yu et al. (1997) | 48/M | Diffuse | Yes | No | NR | |
25 | Chow et al. (1998) | 42/F | Diffuse | Yes | No | No/2 y | |
26 | Omura et al. (1998) | 18/M | Diffuse | Yes | No | No/2 y | |
27 | Stojadinovic et al. (1999) | 63/M | Diffuse | Yes | No | No/20 mo | |
28 | Bemporad et al. (1999) | 37/M | Diffuse | Yes | No | NR | |
29 | Song et al. (1999) | 57/F | Diffuse | Yes | No | NR | |
30 | Lee et al. (2000) | 59/F | Diffuse | Yes | No | No/2 y | |
31 | Kişnişci et al. (2001) | 45/F | Diffuse | Yes | No | No/1 y | |
32 | Klenoff et al. (2001) | 35/NR | Diffuse | Yes | No | No/3 y | |
33 | Church et al. (2003) | 42/M | Diffuse | Yes | No | No/3 y | |
33/M | Diffuse | Yes | No | No/2 y | |||
34 | Heo et al. (2003) | 45/M | NR | NR | NR | NR | |
35 | Kunz et al. (2003) | 13/M | Diffuse | Yes | No | NR/2 y | |
36 | Aoyama et al. (2004) | 33/M | Diffuse | Yes | No | No/2 y | |
37–41 | Kim et al. (2004) | 28/F | Diffuse | NR | NR | NR | |
22/M | Diffuse | NR | NR | NR | |||
47/M | Diffuse | NR | NR | NR | |||
58/F | Diffuse | NR | NR | NR | |||
42 | Tosun et al. (2004) | 60/M | Diffuse | Yes | No | No/7 y | |
43 | Cascone et al. (2005) | 38/F | Localized | Yes | No | No/1 y | |
44 | Izzo et al. (2005) | 67/NR | Localized | Yes | No | No/1 mo | |
45–46 | Stryjakowska et al. (2005) | 36/M | Diffuse | Yes | No | No/3 y | |
36/F | Diffuse | Yes, two times | No | Yes/2 y | |||
47 | Fang et al. (2007) | 44/M | Diffuse | Yes | No | No/20 mo | Radiation suggested, not done |
48–49 | Oda et al. (2007) | 52/M | Diffuse | Yes | No | No/11 mo | With chondroid metaplasia |
67/M | Diffuse | Yes | No | No/7 y and 3 mo | With chondroid metaplasia | ||
50 | Cascone et al. (2008) | 78/M | Localized | Yes | No | No/2 y | |
51 | Chen et al. (2008) | 56/F | Diffuse | Yes | Yes (NR) | No/9 mo | |
52 | Day et al. (2008) | 38/M | Diffuse | Yes | No | No/3 mo | |
53 | Cai, X-Y. et al. (2009) | 21/F | Localized | Yes | No | No/13 mo | With synovial chondromatosis |
54 | Herman et al. (2009) | 36/M | Diffuse | Yes | No | No/11 y | |
55 | Shkoukani et al. (2009) | 74/F | Diffuse | Yes | No | No/NR | |
56 | Gong et al. (2010) | 34/M | Diffuse | Yes | No | No/2 y | TGCT |
57 | Leiggener et al. (2010) | 22/F | Diffuse | Yes | No | No/3 y | |
58 | Cai, J. et al. 2011 | 59/F | Diffuse | Yes | No | No/NR | |
59 | Lu et al. (2011) | 42/M | Diffuse | Yes, two times | No | Yes/44 mo | Recurrence at different location near TMJ |
60–64 | Hoch et al. 2001 | 36-70/3 M, 2 F | Diffuse | Yes | No | No/29 mo avg | With chondroid metaplasia |
65 | Romanach et al. (2011) | 26/M | Diffuse | Yes | Yes (40) | No/18 mo | 20 fractions |
66 | Yoon et al. (2011) | 29/M | Diffuse | Yes | No | No/30 mo | Recurrence in lung at 30 mo, malignant with lung metastasis |
67 | Aimoni et al. (2012) | 80/M | Diffuse | Yes | No | No/18 mo | |
68 | He et al. (2012) | 34/M | Diffuse | Yes | No | No/6 mo | TGCT |
69 | Liu et al. (2012) | 56/F | Diffuse | Yes | Yes (35) | No/2 y 4 mo | 15 fractions |
70 | Tel et al. (2012) | 29/M | Diffuse | Yes | No | No/5 y | |
71 | Wong et al. (2012) | 51/F | Diffuse | Yes | No | NR/1 yr | |
72–73 | Present series (2014) | 53/M | Diffuse | Yes | Yes (45) | No/4 y | 25 fractions |
30/M | Diffuse | Yes | Yes (45) | No/2 y | 25 fractions |
2
Materials and methods
A PubMed query was performed using the keyword combinations of pigmented villonodular synovitis temporomandibular joint and the synonymous term of tenosynovial giant cell tumor temporomandibular joint . Articles in English and non-English (translated) reporting on at least one case of PVNS of the TMJ were included. The references and literature review tables of these articles were also reviewed to ensure all pertinent literature was included. A total of 71 cases were identified in the literature, not including the two cases presented in this report. The details are represented in Table 1 .
No. | Author (date) | Age (y)/ gender | Type of PVNS | Surgery | Radiation (Gy) | Local recurrence/follow-up period | Other notes |
---|---|---|---|---|---|---|---|
1–2 | Lapayowker et al. (1973) | 22/M | Diffuse | Yes | No | No/8 y | |
58/F | Diffuse | Yes | No | NR | |||
3 | Barnard (1975) | 37/M | NR | Conservative approach, biopsy only | No | No change in lesion/6 mo | |
4 | Dinerman and Myers (1977) | 58/F | Diffuse | Yes | No | No/4 y | |
5 | Miyamoto et al. (1977) | 34/M | Diffuse | Yes | No | No/2 y | |
6 | Raibley (1977) | 62/F | Localized | Yes | No | No/5 mo | With synovial chondromatosis |
7 | Makek and Drommer (1978) | 55/F | Localized | Yes | No | No/9 mo | |
8 | Geiger and Pesch (1980) | 50/F | Diffuse | Yes | No | No/4 y | |
9 | Takagi and Ishikawa (1981) | 36/M | Diffuse | Yes, X2 | No | Yes/5 y; No/11 y | |
10 | Gallia et al. (1982) | 47/F | Diffuse | Yes | No | No/2 y | |
11 | Rickert and Shapiro (1982) | 39/F | Diffuse | Yes | No | NR | |
12 | Curtin et al. (1983) | 47/F | Diffuse | Yes | No | NR | |
13 | O’Sullivan et al. (1984) | 61/F | Diffuse | Yes, X3 | Yes, after the fourth recurrence (40) | Yes/X3 in a 7 y period | |
14 | Dawiskiba et al. (1988) | 32/M | Diffuse | NR | No | NR | |
15 | Eisig et al. (1992) | 50/F | Diffuse | Yes | No | No/1 y | |
16 | Syed et al. (1993) | 10/F | Diffuse | Yes | No | NR | |
17 | Franchi et al. (1994) | 59/F | Diffuse | Yes | No | No/1 y | |
18 | Shapiro et al. (1996) | 36/M | Diffuse | Yes | No | No/18 mo | |
19 | Youssef et al. (1996) | 41/F | Localized | Yes | No | No/14 mo | |
20 | Allias-Montmayeur et al. (1997) | 39/F | Localized | Yes | No | No/7 mo | |
21 | Bertoni et al. (1997) | 71/F | Diffuse, metastatic | Yes, x5 | Yes (45) | Yes/X4 in 7 y period | Malignant PVNS, local disease, intracranial extension, and lung metastasis |
22 | Renaga Rubin et al. (1997) | 70/F | Diffuse | Yes | No | No/3 y | |
23 | Tanaka et al. (1997) | 47/M | Diffuse | Yes | No | No/2 y | |
24 | Yu et al. (1997) | 48/M | Diffuse | Yes | No | NR | |
25 | Chow et al. (1998) | 42/F | Diffuse | Yes | No | No/2 y | |
26 | Omura et al. (1998) | 18/M | Diffuse | Yes | No | No/2 y | |
27 | Stojadinovic et al. (1999) | 63/M | Diffuse | Yes | No | No/20 mo | |
28 | Bemporad et al. (1999) | 37/M | Diffuse | Yes | No | NR | |
29 | Song et al. (1999) | 57/F | Diffuse | Yes | No | NR | |
30 | Lee et al. (2000) | 59/F | Diffuse | Yes | No | No/2 y | |
31 | Kişnişci et al. (2001) | 45/F | Diffuse | Yes | No | No/1 y | |
32 | Klenoff et al. (2001) | 35/NR | Diffuse | Yes | No | No/3 y | |
33 | Church et al. (2003) | 42/M | Diffuse | Yes | No | No/3 y | |
33/M | Diffuse | Yes | No | No/2 y | |||
34 | Heo et al. (2003) | 45/M | NR | NR | NR | NR | |
35 | Kunz et al. (2003) | 13/M | Diffuse | Yes | No | NR/2 y | |
36 | Aoyama et al. (2004) | 33/M | Diffuse | Yes | No | No/2 y | |
37–41 | Kim et al. (2004) | 28/F | Diffuse | NR | NR | NR | |
22/M | Diffuse | NR | NR | NR | |||
47/M | Diffuse | NR | NR | NR | |||
58/F | Diffuse | NR | NR | NR | |||
42 | Tosun et al. (2004) | 60/M | Diffuse | Yes | No | No/7 y | |
43 | Cascone et al. (2005) | 38/F | Localized | Yes | No | No/1 y | |
44 | Izzo et al. (2005) | 67/NR | Localized | Yes | No | No/1 mo | |
45–46 | Stryjakowska et al. (2005) | 36/M | Diffuse | Yes | No | No/3 y | |
36/F | Diffuse | Yes, two times | No | Yes/2 y | |||
47 | Fang et al. (2007) | 44/M | Diffuse | Yes | No | No/20 mo | Radiation suggested, not done |
48–49 | Oda et al. (2007) | 52/M | Diffuse | Yes | No | No/11 mo | With chondroid metaplasia |
67/M | Diffuse | Yes | No | No/7 y and 3 mo | With chondroid metaplasia | ||
50 | Cascone et al. (2008) | 78/M | Localized | Yes | No | No/2 y | |
51 | Chen et al. (2008) | 56/F | Diffuse | Yes | Yes (NR) | No/9 mo | |
52 | Day et al. (2008) | 38/M | Diffuse | Yes | No | No/3 mo | |
53 | Cai, X-Y. et al. (2009) | 21/F | Localized | Yes | No | No/13 mo | With synovial chondromatosis |
54 | Herman et al. (2009) | 36/M | Diffuse | Yes | No | No/11 y | |
55 | Shkoukani et al. (2009) | 74/F | Diffuse | Yes | No | No/NR | |
56 | Gong et al. (2010) | 34/M | Diffuse | Yes | No | No/2 y | TGCT |
57 | Leiggener et al. (2010) | 22/F | Diffuse | Yes | No | No/3 y | |
58 | Cai, J. et al. 2011 | 59/F | Diffuse | Yes | No | No/NR | |
59 | Lu et al. (2011) | 42/M | Diffuse | Yes, two times | No | Yes/44 mo | Recurrence at different location near TMJ |
60–64 | Hoch et al. 2001 | 36-70/3 M, 2 F | Diffuse | Yes | No | No/29 mo avg | With chondroid metaplasia |
65 | Romanach et al. (2011) | 26/M | Diffuse | Yes | Yes (40) | No/18 mo | 20 fractions |
66 | Yoon et al. (2011) | 29/M | Diffuse | Yes | No | No/30 mo | Recurrence in lung at 30 mo, malignant with lung metastasis |
67 | Aimoni et al. (2012) | 80/M | Diffuse | Yes | No | No/18 mo | |
68 | He et al. (2012) | 34/M | Diffuse | Yes | No | No/6 mo | TGCT |
69 | Liu et al. (2012) | 56/F | Diffuse | Yes | Yes (35) | No/2 y 4 mo | 15 fractions |
70 | Tel et al. (2012) | 29/M | Diffuse | Yes | No | No/5 y | |
71 | Wong et al. (2012) | 51/F | Diffuse | Yes | No | NR/1 yr | |
72–73 | Present series (2014) | 53/M | Diffuse | Yes | Yes (45) | No/4 y | 25 fractions |
30/M | Diffuse | Yes | Yes (45) | No/2 y | 25 fractions |