- Email: [email protected]
Extra-articular pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature
Int. £ Oral Maxillofac. Surg. 2000; 29:408-415 Printed in Denmark. All rights reserved
Copyright © Munksgaard 2000 tnteraationalJouma] of
Oral&_ Maxofacial Surgery ISSN 0901-5027
Extra-articular pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature
Jong-Ho Lee 1, Young-Youn Kim 1, Byoung-Moo Seo 1, Seung-Hak Baek 2, Jin-Young Choi ~, PilI-Hoon Choung 1, Myung-Jin Kim ~ 1Department of Oral and Maxillofacial Surgery, and 2Department of Orthodontics, College of Dentistry, Seoul National
University, Seoul, Korea
J.-H. Lee, Y.-Y. Kim, R-M. Seo, S.-H. Baek, J.-Y. ChoL P.-H. Choung, M.-J. Kim: Extra-articular pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature. Int. J. Oral Maxillofac. Surg. 2000; 29: 408-415. © Munksgaard, 2000 Abstract. Pigmented villonodular synovitis (PVNS) is a benign proliferative disorder of the synovial tissue, that usually affects the knee, ankle and other major joints. Involvement of the temporomandibular joint is, however, very rare. We describe a new case of recurrent PVNS arising from the temporomandibular joint that was classified as diffuse extra-articular type and was successfully treated surgically. The etiology and clinical features of PVNS are discussed and the previously reported cases in the literature are reviewed.
Pigmented villonodular synovitis (PVNS) is an u n c o m m o n disorder of the synovium that may involve joints, tendon sheaths, and bursae. PVNS of the t e m p o r o m a n d i b u l a r joint (TMJ) is very rare. The earliest description of the disorder, involving a tendon sheath, was attributed to CHASSAIQNAC in 185219. The first reports of articular PVNS are credited to SIMON in 1865 (the focal form) and MOSLER in 1909 (the diffuse form) 19. In 1941, J~tFFE et al. 22 revealed that the various lesions of the hands and joints with their differing gross morphologies are variants of the same disorder. They divided PVNS into 1) the diffuse form that affects the entire synovial m e m b r a n e of a joint or bursa, and 2) the localized form that affects a t e n d o n sheath or a portion of the joint lining. Based on 20 cases, they concluded that: 1) the joint and tendon ab-
normalities were related, 2) the diffuse and focal forms were different manifestations of the same disease process, and 3) these lesions should be included under the collective title "pigmented villonodular synovitis". The occurrence of PVNS in the temporomandibular joint was first described by LAPAYOWKER et a l Y in 1973. EISIG et al. 14, in 1992, reported a case of PVNS of the T M J and reviewed 11 previous cases. Since then, another 11 new cases of this site have been added in the literature. Recently we experienced a destructive PVNS involving the right T M J and parotid area that was diagnosed by magnetic resonance imaging and punch needle biopsy. In this article, a detailed case report and comprehensive literature review of PVNS involving T M J are presented.
Key words: temporomandibular joint; synovitis, pigmented villonodular; PVNS. Accepted for publication 12 June 2000
Case report A 59-year-old Korean lady was referred to our clinic in January 1998 for evaluation and treatment of a right preauricular broadlybulging mass, sized about 4×4 cm. The patient reported that she had had a tumor removed from this area 6 years ago. Previous hospitalization records revealed that the former surgical procedure was a superficial parotidectomy, but that no specific pathologic diagnosis was made at that time. After 3 years, she noticed swelling of the right preauricular area, but considered it not significant because this swelling was small and painless. As time went on, the swelling became so large that she visited our clinic. Clinical examination revealed a non-tender preauricular mass with intact overlying skin (Fig. 1A). Her mouth opening was not limited (maximum interincisal opening, 40 mm) and showed no abnormal TMJ function. The facial nerve was intact but decreased salivary flow was noted from the
P V N S in T M J
409
Fig. 1. A. Clinical photograph demonstrating right-sided preauricular mass. B. Panoramic view showing enlargement of the condylar head. C. Coronal MRI scan shows well-encapsulated mass of low signal intensity involving the right TMJ. D. Bone scan showing hot-spot in the right TMJ.
right Stenson's duct orifice. There was no history or positive findings of joint disease. Plain radiographic studies of the mandible demonstrated enlargement of the condylar process without erosive change (Fig. 1B). T2weiglated coronal MRI scans showed a large lobulated mass of low signal intensity that involved the right TMJ and mandibular condyle, and measured 4.5x4 cm (Fig. 1C). The lesion extended into the region of the right infratemporal fossa. A 99mTc bone scan showed abnormally intense radioisotope uptake at the right TMJ (Fig. 1D). Routine laboratory findings were normal except for high blood glucose (about 130 mg/dl) during fasting. All vital organs were normal but hypertensive. A needle biopsy specimen was obtained percutaneously through the TMJ using a biopsy punch gun under local anesthesia. A punch biopsy and MRI scan confirmed that the lesion was pigmented villonodular synovitis. In Feb. 1998, the patient underwent surgery under general anesthesia with endo-
tracheal intubation. The tumor was approached via the parotidectomy incision. After elevation of a skin flap, the main branches of the facial nerve were isolated. The residual parotid gland and encapsulated mass surrounding the TMJ were identified. The mass was too close to, and in some areas intermingled with, the facial nerve, so it was decided to remove the tumor and the involved facial nerve. The distal branches of the facial nerve (buccal, zygomatic and frontal branches) emitting from the tumor were isolated and marked with silk stitches for later nerve repair. The main mass with the mandibular condyle was delivered after osteotomizing the mandibular ramus. The surgical defect of the mandibular ramuscondylar head was reconstructed using a costochondral graft which was harvested from the 5th rib. The disrupted facial nerve was reconstructed microsurgically with sural nerve segments (Fig. 2). The patient tolerated the procedure very well and was discharged uneventfully.
The total size of the resected specimen was 6.5x4.5×4.5 cm (Fig. 3). The tumor which was attached to the right side of the condylar process measured 4.5×3.5×3.0 cm. It was fairly encapsulated and the color of the cut surface was brown. The villous pattern was not prominent. Microscopic examination revealed proliferation of mononuclear round cells that had plump nuclei with prominent nucleoli and abundant eosinophilic cytoplasm mixed with a number of osteoclast-like giant cells (Fig. 4A). Hemosiderin within the cytoplasm of the histiocytes was focally abundant. On immunohistochemical examination, a-l-antichymotrypsin was focally positive (Fig. 4B), but S-100, vimentin, and lysozyme were negative. The final pathologic diagnosis was pigmented villonodular synovitis. After discharge, electric stimulation therapy and physical therapy were followed to stimulate facial nerve regeneration and to retard atrophy of the facial muscles. A twoyear follow-up has not shown any recurrence
410
Lee et al.
Fig. 3. A. Resected lesion and mandibular condyle. B. On the cut surface, a reddish-brown color was prominent.
Fig. 2. After skin flap elevation, a well-encapsulated mass tightly adherent to the temporomandibular joint was visualized.
of swelling in the temporomandibular joint (Fig. 5A). Maximum interincisal opening was 45 mm with minimal deviation (Fig. 5B). Function of the facial nerve was not fully recovered but was satisfactory. A panoramic radiograph showed remodeling of the costochondral graft (Fig. 5C). There was no sign of recurrence on an MRI scan (Fig. 5D). Discussion
Pigmented villonodular synovitis is a benign proliferative disorder of synovium. JAFFE et al. 22 regarded the synovium of joint, tendon sheath, and bursa as an anatomical unit that could give rise to a common family of lesions in-
cluding pigmented villonodular synovitis, pigmented villonodular tenosynovitis, or pigmented villonodular bursitis depending on the area involved. Other terms used to describe this disorder include xanthoma, xanthogranuloma, fibroxanthoma, chronic hemorrhagic villous synovitis, giant cell fibrohemangioma, fibrohemosideric sarcoma, benign synovioma, sarcoma fusigigantiocellulare, benign polymorphocellular tumor of the synovial membrane, giant cell tumor of tendon sheath, myeloplaxoma, and villous arthritis 19. As already mentioned, two forms are identifiable: a focal or sharply localized form characterized by a solitary pedunculated lesion, and a diffuse form characterized by diffuse involvement of synovial membrane. The diffuse form of this disorder usually involves a single joint and is rarely polyarticular, and is most common in the knee joint (about 80% of cases). Other joints that may be
affected by PVNS, in decreasing order of frequency, include the hip, ankle, and shoulder. The usual case of diffuse PVNS is found in adults of age 20-50, without sex predilection. Diffuse PVNS may invade the joint capsule, soft tissue and bone, and coat adjacent nerves and vessels5. The focal or localized form of PVNS may affect either joints or tendon sheaths. Lesions arising from the synovial tendon sheath (giant cell tumor of tendon sheath) occur most commonly in the digits of the hands and feet. PVNS of the tendon sheath occurs most frequently in the fifth or sixth decades of life. In contrast to the diffuse form, the localized form is more often seen in women. These classification and clinical features are summarized in Table 1. ENZINGER & V~ISS 15 used the term of tenosynovial giant cell tumor. Pigmented villonodular synovitis and tenosynovial giant cell tumor are both re-
Fig. 4. A. Photomicrograph showing multinucleated giant cells (arrow head) and hemosiderin deposits (arrow) (hematoxylin-eosin stain, original magnification × 150). B. Immunohistochemical stain for c~-1-antichymotrypsin (original magnification × 100).
P V N S in T M J
411
Fig. 5. A. Clinical photograph of 2-year follow-up. B. Mandibular opening with minimal deviation. C. Follow-up panoramic view showed remodeling of costochondral graft. D. Follow-up MRI scan at 2 years demonstrated no recurrence.
garded as a c o m m o n family of synovial lesions because of their histologic similarity. These authors classified tenosy-
novial giant cell tumor into two types; diffuse or localized. Diffuse tenosynovial giant cell tumor is also referred to as
proliferative synovitis, florid synovitis, extra-articular pigmented villonodular synovitis, and pigmented villonodular
Table 1. Summary of classification of PVNS
Diffuse
Localized
General terminology
Pigmented villonodular synovitis
Pigmented villonodular bursitis
Pigmented villonodular tenosynovitis
Localized nodular synovitis
Primary origin
Intra-articular
Extra-articular (bursa)
Tendon sheath
Articular
Predominant age
2040
Under 40
40-60
40-60
Predominant gender
Male=Female
Females affected slightly more than males
Female
Male
Site typically affected
Knee joint
Knee joint
Digits of hands and feet
Knee joint
Diffuse tenosynovial giant cell tumor
Localized tenosynovial giant cell tumor
Terminology of ENZINGER& WEISS15
412
Lee et al.
0
0
0
0
~"
0
0
z
z
0
~
~
0
~,z
Z
~Z
Z
'm
..
,o
o
~.,~
o~ Z
¢1.~"¢1
~
z
. ~
o
0.~
~
~
"*~
"
N~,
~o~'== ~
o
oo
. o~
o
~-~ ~
•~ o
•~
.o
~
~.~
-
~-~
~
~'~
~o~
•
Z
.o
.= o . ~ 9 Z
e-i
~ ~ ~x
Z ,--a r~
~3
Z
,--i
•
~
~o
<
~
z 0 m
e,,l
==
< ~Z
o~
¢N
¢~
,~-
t¢3
o
P V N S in T M J
~
Z
Z
Z
°
Z
Z
Z
Z
0
e.
O O
O
-r, ~6 Z
O
..~ ~.~ o
~..~
~5
z
z O
-°.o
~..~ m
=eN
.o e~
~=
g
~
,-.M
~]
e~ ,.O ,- o ' ~ O ~ ,X~ ~ ' ~ ¢~
~
~
>
.o= o
m o ~.~ ~ . ~ "~ ~ . ~ -~
"o
b"
o~
O
~.~
~
~.~
g
O .,--~
~.~ o=
=
~ ~.~
.o
O
r~
o.
~N N .N
,.o ~0
~ .~ =.~
~O ' . ~o ~ = . ~
~2
o
~2
i
o~ ~4
g~
N
N 0 0
,--A
o .~,
z
¢,1
e~ 0 • . Q)
[.., r..) O
0
0
~)
0
O
413
bursitis. Localized tenosynovial giant cell tumor is a synonym of nodular tenosynovitis and giant cell tumor of the tendon sheath. According to this classification, OMURA et al. 28 reported one case of diffuse tenosynovial giant cell tumor of the TMJ and described this as the first case of diffuse tenosynovial giant cell tumor involving the TMJ. But in our opinion, their case is one of PVNS (extra-articular type) because ENZINGER & VVEISS 15 indicated that diffuse tenosynovial giant cell tumor is the synonym of extra-articular PVNS. Generally and historically the term of PVNS was used in the context of a collective term, so in our opinion, it was not wise to use the terminology according to the origin (intra-articular or extra-articular) because intra-articular lesions may extend to the extra-articular area, and the reverse is possible. As a similar entity, one case of nodular synovitis involving the TMJ was reported in 197826. Histologic findings of this case were similar to PVNS, with the exception that the inner joint lining was smooth, and no hemosiderin was deposited near the surface. According to MAKEK & D R O M M E R 26, the distinction between villonodular synovitis and localized nodular synovitis and recognition of these two clinical entities of nearly similar histomorphological pattern is of great importance, as they differ in clinical course, treatment and prognosis. The etiology of PVNS is controversial. Four prevalent theories concerning the etiology are 1) a disturbance of lipid metabolism, 2) a response to blood or blood products, 3) an inflammatory response to an unknown nontraumatic irritant, and 4) a benign neoplasm of synovial, vascular, or fibrohistiocytic origin 19. The two most widely accepted theories are either a chronic inflammatory process or a benign neoplasia, although JAF~ et al. 22 originally suggested that this lesion was an inflammatory response based on the histologic findings of hyperplastic stromal cells with phagocytic qualities, abundant collagen, and hyalinization with progressive fibrosis. Some investigators prefer 'tumor' to 'synovitis'. Proponents of a neoplastic origin have demonstrated monoclonality of the cells6, chromosomal abnormalities 6 and metastatic cases 3. According to PAMMER et al. 31, however, giant cells of PVNS and other bone and soft tumors are of reactive, rather than neoplastic origin.
414
L e e et al.
Recent cell marker studies have indicated that the tumor cells are closely related to monocytes and macrophages 23. According to JozsA 23, the mononuclear cells were found to stain positively with anti-lysozyme, anti-a- 1-antitrypsin, anti-a-l-antichymotrypsin and antifibronection, and multinuclear giant cells contained lysozyme, a-l-antitrypsin and a-l-antichymotrypsin. Immunohistochemical study of our case showed that a-l-antichymotrypsin was focally positive. PVNS rarely affects the temporomandibular joint. Since the first report of LAPAYOWKER et al. 25, a total of 22 cases involving the TMJ have been reported in the literature. These cases are summarized in Table 2. The average age of the cases was 44.6 years (ranged from 10 to 70 years old); 12 were women and 10 were men. Clinically, the presentation of this lesion of the TMJ was a progressive preauricular swelling. Pain was not significant. Clinical symptoms of temporomandibular disorders such as clicking, limitation of mouth opening, malocclusion, and painful mastication were usually present. Other symptoms included hearing loss, otalgia, diplopia, and vomiting. Preoperatively the lesion was usually diagnosed as a parotid tumor. Surgical specimens reveal that the synovium has a mossy or nodular texture, spongy cut surface, and a rusty, red-brown, or yellow-brown color. Microscopically, the synovium is composed of a fingerlike or rounded mass of fibrous stroma covered by hyperplastic lining cells. Multinucleated giant cells in varying numbers are a characteristic feature. Hemosiderin pigment is present as well and is usually found concentrated in the stromal cells. This hemosiderin is a breakdown product of hemoglobin, and its presence signifies old hemorrhage 12. Panoramic radiography of our case showed the enlargement of the condylar process. Usually, plain radiographic findings include lytic lesions involving the condyle and glenoid fossa, erosion of the juxta-articular bone, and massive destruction of the TMJ. Frequently the lesions extend into the temporal bone and even the middle cranial fossa in seven references. These findings are more evident in CT or MRI. Especially in MRI, the presence of hemosiderin results in the most characteristic findings. In particular, the ferromagnetic properties of hemosiderin cause shortening
of both T1 and T2 relaxation time. This effect is likely to be concentration-dependent, and when PVNS tissue contains enough hemosiderin, it appears characteristically dark on all pulse sequences 5. M R I also shows a cyst erosion on the temporal bone and degenerative changes on the mandibular condyle. Magnetic resonance imaging in our case showed the characteristic appearance of PVNS and clearly delineated the extent of the lesion. Thus, it was very useful in preoperative planning aimed at obtaining adequate surgical margins. The pathogenesis of bone erosion is uncertain. The most favored postulate is that of CHUNG & JANES s, who proposed that synovial overgrowth and joint effusion cause high intra-articular pressure; in turn, focal areas of demineralization eventuate in cyst formation followed by fracture of the cyst walls and invasion of tumor. BREIMER& FREIB~R6ZR4, however, stated that the mechanism of bone destruction is compression of the soft tissue mass. DARLiNG et al. l° showed that the PVNS synovium expresses metalloproteinases collagenase (MMP-1) and stromelysin (MMP-3) in varying amounts. These results suggest that collagenase and stromelysin may be among the mediators of cartilage and bone loss that can occur in PVNS. Radical excision is the treatment of choice for PVNS, especially in the case of extra-articular type. Although wide excision is the best choice for local control of disease, it obviously implies significant morbidity, especially for lesions located adjacent to major joints. In our case, we decided to excise widely and to reconstruct the condyle with a costochondral graft. This disease is said to be radiosensitive by some authors, but the results were difficult to assess in cases involving the TMJ. O'SULLIVANet al. 29 reported the results of radiotherapy on 14 cases of pigmented villonodular synovitis. Their results showed that moderate dose radiotherapy (35 Gy in 15 fractions) is an effective modality in the treatment of a subset of cases with this rare condition. The most common complication after surgerY is facial paralysis. This occurred in three cases. However, facial nerve function recovery was almost complete in most cases. Although it is difficult to predict the recurrence rate of PVNS in this location because of the short follow-up periods,
our literature review shows that the recurrence rate was 9% (2 out of 22 cases). This rate is lower than that of other joints, the recurrence rates of which range from 21% 21 to 48% 35. Recurrence is presumably due to incomplete excision 19. Cases of spontaneous regression have been reported 2°. Malignant type or malignant transformation of pigmented villonodular synovitis is very rare and its existence may be debatable. BERTONI et al. 3 reported eight cases of malignant PVNS. According to them, important histologic features of malignancy were 1) a nodular, solid infiltrative pattern of the indistinct borders, 2) large, plump, round or oval cells with deep eosinophilic cytoplasm and indistinct borders, 3) large nuclei with prominent nucleoli, and 4) necrotic areas. In the region of the TMJ, one case of malignant PVNS was reported 3. This patient had four recurrences during a 7-year period. The third recurrence, deemed inoperable, was treated with 4,500 rad of radiation therapy. But the patient had pulmonary metastasis 4 years after radiation therapy. She died after the detection of metastasis, 17 years after the first diagnosis. KALIL & UNN124 reported one Case of malignant PVNS of the ankle. This patient showed lymph node metastasis. In summary, pigmented villonodular synovitis is indeed a rare lesion of the temporomandibular joint. However, we recommend that PVNS be included in the differential considerations when preauricular swelling with trismus is present. More frequent use of M R I might help diagnosis of this disease. Careful preoperative planning and complete resection are the factors that lead to the optimal treatment of PVNS.
References
1. BARNARDJDW. Pigmented villonodular synovitis in the temporomandibular joint: a case report. Br J Oral Surg 1975: 13: 183-7. 2. BEMrO~D JA, CI~ALOUPKAJC, PUT~N CM, et al. Pigmented villonodular synovitis of the temporomandibular joint: diagnostic imaging and endovascular therapeutic embolization of a rare head and neck tumor. AJNR Am J Neuroradiol 1999: 20: 159-62. 3. BERTONIF~ UNNI KK, BEABOUTJW, SIM FH. Malignant giant cell tumor of the tendon sheaths and joints (malignant pigmented villonodular synovitis). Am J Surg Pathol 1997: 21: 153-63.
P V N S in T M J 4. BREIMER CW, FREIBERGER RH. Bone lesions associated with villonodular synovitis. Am J Roentgenol 1958: 79:618 29. 5. BRAVOSM, WINALSKICS, WEISSMANBN. Pigmented villonodular synovitis. Radiol Clin North Am 1996: 34: 31156. 6. CHOONG PF, WILLEN H, NILBERT M, MERTENS F, MANDAHL N, CARLEN B, RYDHOLM A. Pigmented villonodular synovitis. Monoclonality and metastasis - a case for neoplastic origin? Acta Orthop Scand 1995: 66: 64-8. 7. CHOW LT, KUMTASM, KING WW. Extraarticular pigmented villonodular synovitis of the temporomandibular joint. J Laryngol Otol 1998: 112: 182-5. 8. CHUNG SMK, JANES JM. Diffuse pigmented villonodular synovitis of the hip joint. Review of the literature and report of four cases. J Bone Joint Surg 1965: 47A: 293 303. 9. CURTIN HI), WILLIAMSR, GALLA L, MEYERS EN. Pigmented villonodular synovitis of the temporomandibular joint. Comput Radiol 1983: 7:257 60. 10. DARLING JM, GLIMCHER LH, SHORTKROFF S, ALBANO B, GRAVALLESE EM. Expression of metalloproteinases in pigmented villonodular synovitis. Hum Pathol 1994: 25: 825-30. 11. D1NERMAN WS, MYERS EN. Pigmented villonodular tenosynovitis of the temporomandibular joint. Trans Am Acad Opthalmol Otol 1977: 84: 132-5. 12. DORWART RH, GENANT HK, JOHNSTON WH, MORRIS JM. Pigmented villonodular synovitis of synovial joints: clinical, pathologic, and radiologic features. AJR Am J Roentgenol 1984: 143: 87785. 13. DWAISKIBA S, ERIKSSON L, ELNER A, JOHANSEN CC, HANSSON LG, ~V'ESTESSON PL. Diffuse pigmented villonodnlar synovitis of the temporomandibular joint diagnosed by fine needle aspiration cytology. Diagn Cytol 1989: 5: 301-4. 14. EISlG S, DORFMAN HI), CUSAMANO RJ, KANTROWITZ AB. Pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature. Oral Surg Oral Med Oral Pathol 1992: 73: 328-33. 15. ENZINGER FM, V@ISSSW Soft tissue tumors. St Louis: C. V. Mosby, 1995:735 55. 16. FRANCHI A, FROSINI P, SANTORO R. Pigmented villonodular synovitis of the temporomandibular joint: report of a case. J Laryngol Otol 1994: 108:166 7. 17. GALLIA LJ, JOHNSON JT, MYERS EN. Pigmented villonodular synovitis of the tern-
poromandibular joint: a case report. Otolaryngol Head Neck Surg 1982: 90:691 5. 18. GEIGER S, PESCH HJ. Synovitis pigmentosa villonodularis, eine seltene Kiefergelenkerkrankung. Fortschr Kiefer Gesichtschir 1980: 25: 29-31. 19. GOLDMAN AB, DICARLO EE Pigmented villonodular synovitis: diagnosis and differential diagnosis. Radiol Clin North Am 1988: 26: 327-47. 20. GRANOWlTZ SP, D'ANTONIO J, MANKIN HL. The pathogenesis and long-term end results of pigmented villonodular synovitis. Clin Orthop 1976: 114: 335-51. 21. GREENFIELD MM, WALLACE KM. Pigmented villonodular synovitis. Radiol 1950: 54: 350-6. 22. JAFFE HL, LICHTENSTEIN L, SUTRO CJ. Pigmented villonodular synovitis, bursitis and tenosynovifis. Arch Pathol 1941: 31: 731 65. 23. JOZSA L. Immunohistochemical characterization of pigmented villonodular synovitis. Zentralbl Pathol 1992: 138: 119 23. 24. KALIL RK, UNNI KK. Malignancy in pigmented villonodular synovitis. Skeletal Radio1 1998: 27: 392-5. 25. LAPAYOWKER MS, MILLER WT, LEVY WM, HARWlCK RD. PVNS of temporomandibular joint. Radiology 1973: 108: 313 6. 26. MAKEKM, DRUMMERR. Localized nodular synovitis of the temporomandibular joint. A case report. J Maxillofac Surg 1978: 6: 302-5. 27. MIYAMOTO Y, TANI T, HAMAYA K. Pigmented villonodular synovifis of the temporomandibular joint: a case report. Hast Reconstr Surg 1977: 59: 283-6. 28. OMURAS, MIZUKI N, BUKAWAH, FUJITA K. Diffuse variant tenosynovial giant cell tumor of the temporomandibular joint: report of a case. J Oral Maxillofac Surg 1998: 56: 991-6. 29. O'SULLIVAN B, CUMMINGSB, CATTON C, BELL R, DAVIS A, FORNASIER V, GOLDBERG R. Outcome following radiation treatment for high-risk pigmented villonodular synovitis. Int J Radiat Oncol Biol Plays 1995: 32: 777-86. 30. O'SULLIVAN T J, ALPORT EC, WHISTON HG. Pigmented villonodular synovitis of the temporomandibular joint. J Otolaryngol 1984: 13: 123-6. 31. PAMMERJ, WENINGERW, HULLA H, MAZAL P, HORVAT R. Expression of regulatory apoptotic proteins in peripheral giant cell granulomas and lesions containing osteoclast-like giant cells. J Oral Pathol Med 1998: 27: 267-71.
415
32. RAIBLEY SO. Villonodular synovitis with chondromatosis. Oral Surg Oral Med Oral Pathol 1977: 44: 279-84. 33. RENAGA RUBIN I, SALAVER GIRONA A, VASQUEZ RODRIGUEZ A, ANMELLA VALMANYA J. Pigmented villonodular synovitis of the temporomandibular joint. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997: 84:459 60. 34. RICKERT RR, SHAPIRO MJ. Pigmented villonodular synovitis of the temporomandibular joint. Otolaryngol Head Neck Surg 1982: 90: 668-70. 35. SHAPIRO S, KESSLER S, MCMENOMEY SO. Pathologic quiz case 1. Temporomandibular pigmented villonodular synovitis. Arch Otolaryngol Head Neck Surgery 1996: 122: 194-96. 36. STOJADINOVIC S, RE1NERT S, WILDFORSTER U, JUNDT G, MACHTENS E. Pigmentierte villonodul/ire Synovitis des Kiefergelenks mit Einbruch in die mittlere Schiidelgrube. Mund Kiefer Gesichtschir 1998: 2:279 81. 37. SYED A, VAN HASSELT CA, TO KE Pigmented villonodular synovitis of the temporomandibular joint. J Laryngol Otol 1993: 107: 8534. 38. TAKAGI M, ISHIKAWA G. Simultaneous villonodular synovitis and synovial chondromatosis of the temporomandibular joint: report of a case. J Oral Surg 1981: 39: 699-701. 39. TANAKAK, SUZUKI M, NAMEKI H, SUGIYAMA H. Pigmented villonodular synovitis of the temporomandibular joint. Arch Otolarygol Head Neck Surg 1997: 123: 536-9. 40. YOUSSEF RE, ROSZKOWSKI M J, RICHTER KJ. Pigmented villonodular synovitis of the temporomandibuar joint. J Oral Maxillofac Surg 1996: 54: 224-7. 41. Yu GH, STAERKELGA, KERSHISNIKMM, VARMA DG. Fine-needle aspiration of pigmented villonodular synovitis of the temporomandibular joint masquerading as a primary parotid gland lesion. Diagn Cytopathol 1997: 16: 47-50.
Address: Dr. Jong-Ho Lee Department of Oral & Maxillofacial Surgery Seoul National University Hospital 28 Yeon-gun-dong, Chong-no-gu Seoul South Korea Tel." +82 2 760 3813 Fax." +82 2 766 4948 e-mail: [email protected]
Copyright © Munksgaard 2000 tnteraationalJouma] of
Oral&_ Maxofacial Surgery ISSN 0901-5027
Extra-articular pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature
Jong-Ho Lee 1, Young-Youn Kim 1, Byoung-Moo Seo 1, Seung-Hak Baek 2, Jin-Young Choi ~, PilI-Hoon Choung 1, Myung-Jin Kim ~ 1Department of Oral and Maxillofacial Surgery, and 2Department of Orthodontics, College of Dentistry, Seoul National
University, Seoul, Korea
J.-H. Lee, Y.-Y. Kim, R-M. Seo, S.-H. Baek, J.-Y. ChoL P.-H. Choung, M.-J. Kim: Extra-articular pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature. Int. J. Oral Maxillofac. Surg. 2000; 29: 408-415. © Munksgaard, 2000 Abstract. Pigmented villonodular synovitis (PVNS) is a benign proliferative disorder of the synovial tissue, that usually affects the knee, ankle and other major joints. Involvement of the temporomandibular joint is, however, very rare. We describe a new case of recurrent PVNS arising from the temporomandibular joint that was classified as diffuse extra-articular type and was successfully treated surgically. The etiology and clinical features of PVNS are discussed and the previously reported cases in the literature are reviewed.
Pigmented villonodular synovitis (PVNS) is an u n c o m m o n disorder of the synovium that may involve joints, tendon sheaths, and bursae. PVNS of the t e m p o r o m a n d i b u l a r joint (TMJ) is very rare. The earliest description of the disorder, involving a tendon sheath, was attributed to CHASSAIQNAC in 185219. The first reports of articular PVNS are credited to SIMON in 1865 (the focal form) and MOSLER in 1909 (the diffuse form) 19. In 1941, J~tFFE et al. 22 revealed that the various lesions of the hands and joints with their differing gross morphologies are variants of the same disorder. They divided PVNS into 1) the diffuse form that affects the entire synovial m e m b r a n e of a joint or bursa, and 2) the localized form that affects a t e n d o n sheath or a portion of the joint lining. Based on 20 cases, they concluded that: 1) the joint and tendon ab-
normalities were related, 2) the diffuse and focal forms were different manifestations of the same disease process, and 3) these lesions should be included under the collective title "pigmented villonodular synovitis". The occurrence of PVNS in the temporomandibular joint was first described by LAPAYOWKER et a l Y in 1973. EISIG et al. 14, in 1992, reported a case of PVNS of the T M J and reviewed 11 previous cases. Since then, another 11 new cases of this site have been added in the literature. Recently we experienced a destructive PVNS involving the right T M J and parotid area that was diagnosed by magnetic resonance imaging and punch needle biopsy. In this article, a detailed case report and comprehensive literature review of PVNS involving T M J are presented.
Key words: temporomandibular joint; synovitis, pigmented villonodular; PVNS. Accepted for publication 12 June 2000
Case report A 59-year-old Korean lady was referred to our clinic in January 1998 for evaluation and treatment of a right preauricular broadlybulging mass, sized about 4×4 cm. The patient reported that she had had a tumor removed from this area 6 years ago. Previous hospitalization records revealed that the former surgical procedure was a superficial parotidectomy, but that no specific pathologic diagnosis was made at that time. After 3 years, she noticed swelling of the right preauricular area, but considered it not significant because this swelling was small and painless. As time went on, the swelling became so large that she visited our clinic. Clinical examination revealed a non-tender preauricular mass with intact overlying skin (Fig. 1A). Her mouth opening was not limited (maximum interincisal opening, 40 mm) and showed no abnormal TMJ function. The facial nerve was intact but decreased salivary flow was noted from the
P V N S in T M J
409
Fig. 1. A. Clinical photograph demonstrating right-sided preauricular mass. B. Panoramic view showing enlargement of the condylar head. C. Coronal MRI scan shows well-encapsulated mass of low signal intensity involving the right TMJ. D. Bone scan showing hot-spot in the right TMJ.
right Stenson's duct orifice. There was no history or positive findings of joint disease. Plain radiographic studies of the mandible demonstrated enlargement of the condylar process without erosive change (Fig. 1B). T2weiglated coronal MRI scans showed a large lobulated mass of low signal intensity that involved the right TMJ and mandibular condyle, and measured 4.5x4 cm (Fig. 1C). The lesion extended into the region of the right infratemporal fossa. A 99mTc bone scan showed abnormally intense radioisotope uptake at the right TMJ (Fig. 1D). Routine laboratory findings were normal except for high blood glucose (about 130 mg/dl) during fasting. All vital organs were normal but hypertensive. A needle biopsy specimen was obtained percutaneously through the TMJ using a biopsy punch gun under local anesthesia. A punch biopsy and MRI scan confirmed that the lesion was pigmented villonodular synovitis. In Feb. 1998, the patient underwent surgery under general anesthesia with endo-
tracheal intubation. The tumor was approached via the parotidectomy incision. After elevation of a skin flap, the main branches of the facial nerve were isolated. The residual parotid gland and encapsulated mass surrounding the TMJ were identified. The mass was too close to, and in some areas intermingled with, the facial nerve, so it was decided to remove the tumor and the involved facial nerve. The distal branches of the facial nerve (buccal, zygomatic and frontal branches) emitting from the tumor were isolated and marked with silk stitches for later nerve repair. The main mass with the mandibular condyle was delivered after osteotomizing the mandibular ramus. The surgical defect of the mandibular ramuscondylar head was reconstructed using a costochondral graft which was harvested from the 5th rib. The disrupted facial nerve was reconstructed microsurgically with sural nerve segments (Fig. 2). The patient tolerated the procedure very well and was discharged uneventfully.
The total size of the resected specimen was 6.5x4.5×4.5 cm (Fig. 3). The tumor which was attached to the right side of the condylar process measured 4.5×3.5×3.0 cm. It was fairly encapsulated and the color of the cut surface was brown. The villous pattern was not prominent. Microscopic examination revealed proliferation of mononuclear round cells that had plump nuclei with prominent nucleoli and abundant eosinophilic cytoplasm mixed with a number of osteoclast-like giant cells (Fig. 4A). Hemosiderin within the cytoplasm of the histiocytes was focally abundant. On immunohistochemical examination, a-l-antichymotrypsin was focally positive (Fig. 4B), but S-100, vimentin, and lysozyme were negative. The final pathologic diagnosis was pigmented villonodular synovitis. After discharge, electric stimulation therapy and physical therapy were followed to stimulate facial nerve regeneration and to retard atrophy of the facial muscles. A twoyear follow-up has not shown any recurrence
410
Lee et al.
Fig. 3. A. Resected lesion and mandibular condyle. B. On the cut surface, a reddish-brown color was prominent.
Fig. 2. After skin flap elevation, a well-encapsulated mass tightly adherent to the temporomandibular joint was visualized.
of swelling in the temporomandibular joint (Fig. 5A). Maximum interincisal opening was 45 mm with minimal deviation (Fig. 5B). Function of the facial nerve was not fully recovered but was satisfactory. A panoramic radiograph showed remodeling of the costochondral graft (Fig. 5C). There was no sign of recurrence on an MRI scan (Fig. 5D). Discussion
Pigmented villonodular synovitis is a benign proliferative disorder of synovium. JAFFE et al. 22 regarded the synovium of joint, tendon sheath, and bursa as an anatomical unit that could give rise to a common family of lesions in-
cluding pigmented villonodular synovitis, pigmented villonodular tenosynovitis, or pigmented villonodular bursitis depending on the area involved. Other terms used to describe this disorder include xanthoma, xanthogranuloma, fibroxanthoma, chronic hemorrhagic villous synovitis, giant cell fibrohemangioma, fibrohemosideric sarcoma, benign synovioma, sarcoma fusigigantiocellulare, benign polymorphocellular tumor of the synovial membrane, giant cell tumor of tendon sheath, myeloplaxoma, and villous arthritis 19. As already mentioned, two forms are identifiable: a focal or sharply localized form characterized by a solitary pedunculated lesion, and a diffuse form characterized by diffuse involvement of synovial membrane. The diffuse form of this disorder usually involves a single joint and is rarely polyarticular, and is most common in the knee joint (about 80% of cases). Other joints that may be
affected by PVNS, in decreasing order of frequency, include the hip, ankle, and shoulder. The usual case of diffuse PVNS is found in adults of age 20-50, without sex predilection. Diffuse PVNS may invade the joint capsule, soft tissue and bone, and coat adjacent nerves and vessels5. The focal or localized form of PVNS may affect either joints or tendon sheaths. Lesions arising from the synovial tendon sheath (giant cell tumor of tendon sheath) occur most commonly in the digits of the hands and feet. PVNS of the tendon sheath occurs most frequently in the fifth or sixth decades of life. In contrast to the diffuse form, the localized form is more often seen in women. These classification and clinical features are summarized in Table 1. ENZINGER & V~ISS 15 used the term of tenosynovial giant cell tumor. Pigmented villonodular synovitis and tenosynovial giant cell tumor are both re-
Fig. 4. A. Photomicrograph showing multinucleated giant cells (arrow head) and hemosiderin deposits (arrow) (hematoxylin-eosin stain, original magnification × 150). B. Immunohistochemical stain for c~-1-antichymotrypsin (original magnification × 100).
P V N S in T M J
411
Fig. 5. A. Clinical photograph of 2-year follow-up. B. Mandibular opening with minimal deviation. C. Follow-up panoramic view showed remodeling of costochondral graft. D. Follow-up MRI scan at 2 years demonstrated no recurrence.
garded as a c o m m o n family of synovial lesions because of their histologic similarity. These authors classified tenosy-
novial giant cell tumor into two types; diffuse or localized. Diffuse tenosynovial giant cell tumor is also referred to as
proliferative synovitis, florid synovitis, extra-articular pigmented villonodular synovitis, and pigmented villonodular
Table 1. Summary of classification of PVNS
Diffuse
Localized
General terminology
Pigmented villonodular synovitis
Pigmented villonodular bursitis
Pigmented villonodular tenosynovitis
Localized nodular synovitis
Primary origin
Intra-articular
Extra-articular (bursa)
Tendon sheath
Articular
Predominant age
2040
Under 40
40-60
40-60
Predominant gender
Male=Female
Females affected slightly more than males
Female
Male
Site typically affected
Knee joint
Knee joint
Digits of hands and feet
Knee joint
Diffuse tenosynovial giant cell tumor
Localized tenosynovial giant cell tumor
Terminology of ENZINGER& WEISS15
412
Lee et al.
0
0
0
0
~"
0
0
z
z
0
~
~
0
~,z
Z
~Z
Z
'm
..
,o
o
~.,~
o~ Z
¢1.~"¢1
~
z
. ~
o
0.~
~
~
"*~
"
N~,
~o~'== ~
o
oo
. o~
o
~-~ ~
•~ o
•~
.o
~
~.~
-
~-~
~
~'~
~o~
•
Z
.o
.= o . ~ 9 Z
e-i
~ ~ ~x
Z ,--a r~
~3
Z
,--i
•
~
~o
<
~
z 0 m
e,,l
==
< ~Z
o~
¢N
¢~
,~-
t¢3
o
P V N S in T M J
~
Z
Z
Z
°
Z
Z
Z
Z
0
e.
O O
O
-r, ~6 Z
O
..~ ~.~ o
~..~
~5
z
z O
-°.o
~..~ m
=eN
.o e~
~=
g
~
,-.M
~]
e~ ,.O ,- o ' ~ O ~ ,X~ ~ ' ~ ¢~
~
~
>
.o= o
m o ~.~ ~ . ~ "~ ~ . ~ -~
"o
b"
o~
O
~.~
~
~.~
g
O .,--~
~.~ o=
=
~ ~.~
.o
O
r~
o.
~N N .N
,.o ~0
~ .~ =.~
~O ' . ~o ~ = . ~
~2
o
~2
i
o~ ~4
g~
N
N 0 0
,--A
o .~,
z
¢,1
e~ 0 • . Q)
[.., r..) O
0
0
~)
0
O
413
bursitis. Localized tenosynovial giant cell tumor is a synonym of nodular tenosynovitis and giant cell tumor of the tendon sheath. According to this classification, OMURA et al. 28 reported one case of diffuse tenosynovial giant cell tumor of the TMJ and described this as the first case of diffuse tenosynovial giant cell tumor involving the TMJ. But in our opinion, their case is one of PVNS (extra-articular type) because ENZINGER & VVEISS 15 indicated that diffuse tenosynovial giant cell tumor is the synonym of extra-articular PVNS. Generally and historically the term of PVNS was used in the context of a collective term, so in our opinion, it was not wise to use the terminology according to the origin (intra-articular or extra-articular) because intra-articular lesions may extend to the extra-articular area, and the reverse is possible. As a similar entity, one case of nodular synovitis involving the TMJ was reported in 197826. Histologic findings of this case were similar to PVNS, with the exception that the inner joint lining was smooth, and no hemosiderin was deposited near the surface. According to MAKEK & D R O M M E R 26, the distinction between villonodular synovitis and localized nodular synovitis and recognition of these two clinical entities of nearly similar histomorphological pattern is of great importance, as they differ in clinical course, treatment and prognosis. The etiology of PVNS is controversial. Four prevalent theories concerning the etiology are 1) a disturbance of lipid metabolism, 2) a response to blood or blood products, 3) an inflammatory response to an unknown nontraumatic irritant, and 4) a benign neoplasm of synovial, vascular, or fibrohistiocytic origin 19. The two most widely accepted theories are either a chronic inflammatory process or a benign neoplasia, although JAF~ et al. 22 originally suggested that this lesion was an inflammatory response based on the histologic findings of hyperplastic stromal cells with phagocytic qualities, abundant collagen, and hyalinization with progressive fibrosis. Some investigators prefer 'tumor' to 'synovitis'. Proponents of a neoplastic origin have demonstrated monoclonality of the cells6, chromosomal abnormalities 6 and metastatic cases 3. According to PAMMER et al. 31, however, giant cells of PVNS and other bone and soft tumors are of reactive, rather than neoplastic origin.
414
L e e et al.
Recent cell marker studies have indicated that the tumor cells are closely related to monocytes and macrophages 23. According to JozsA 23, the mononuclear cells were found to stain positively with anti-lysozyme, anti-a- 1-antitrypsin, anti-a-l-antichymotrypsin and antifibronection, and multinuclear giant cells contained lysozyme, a-l-antitrypsin and a-l-antichymotrypsin. Immunohistochemical study of our case showed that a-l-antichymotrypsin was focally positive. PVNS rarely affects the temporomandibular joint. Since the first report of LAPAYOWKER et al. 25, a total of 22 cases involving the TMJ have been reported in the literature. These cases are summarized in Table 2. The average age of the cases was 44.6 years (ranged from 10 to 70 years old); 12 were women and 10 were men. Clinically, the presentation of this lesion of the TMJ was a progressive preauricular swelling. Pain was not significant. Clinical symptoms of temporomandibular disorders such as clicking, limitation of mouth opening, malocclusion, and painful mastication were usually present. Other symptoms included hearing loss, otalgia, diplopia, and vomiting. Preoperatively the lesion was usually diagnosed as a parotid tumor. Surgical specimens reveal that the synovium has a mossy or nodular texture, spongy cut surface, and a rusty, red-brown, or yellow-brown color. Microscopically, the synovium is composed of a fingerlike or rounded mass of fibrous stroma covered by hyperplastic lining cells. Multinucleated giant cells in varying numbers are a characteristic feature. Hemosiderin pigment is present as well and is usually found concentrated in the stromal cells. This hemosiderin is a breakdown product of hemoglobin, and its presence signifies old hemorrhage 12. Panoramic radiography of our case showed the enlargement of the condylar process. Usually, plain radiographic findings include lytic lesions involving the condyle and glenoid fossa, erosion of the juxta-articular bone, and massive destruction of the TMJ. Frequently the lesions extend into the temporal bone and even the middle cranial fossa in seven references. These findings are more evident in CT or MRI. Especially in MRI, the presence of hemosiderin results in the most characteristic findings. In particular, the ferromagnetic properties of hemosiderin cause shortening
of both T1 and T2 relaxation time. This effect is likely to be concentration-dependent, and when PVNS tissue contains enough hemosiderin, it appears characteristically dark on all pulse sequences 5. M R I also shows a cyst erosion on the temporal bone and degenerative changes on the mandibular condyle. Magnetic resonance imaging in our case showed the characteristic appearance of PVNS and clearly delineated the extent of the lesion. Thus, it was very useful in preoperative planning aimed at obtaining adequate surgical margins. The pathogenesis of bone erosion is uncertain. The most favored postulate is that of CHUNG & JANES s, who proposed that synovial overgrowth and joint effusion cause high intra-articular pressure; in turn, focal areas of demineralization eventuate in cyst formation followed by fracture of the cyst walls and invasion of tumor. BREIMER& FREIB~R6ZR4, however, stated that the mechanism of bone destruction is compression of the soft tissue mass. DARLiNG et al. l° showed that the PVNS synovium expresses metalloproteinases collagenase (MMP-1) and stromelysin (MMP-3) in varying amounts. These results suggest that collagenase and stromelysin may be among the mediators of cartilage and bone loss that can occur in PVNS. Radical excision is the treatment of choice for PVNS, especially in the case of extra-articular type. Although wide excision is the best choice for local control of disease, it obviously implies significant morbidity, especially for lesions located adjacent to major joints. In our case, we decided to excise widely and to reconstruct the condyle with a costochondral graft. This disease is said to be radiosensitive by some authors, but the results were difficult to assess in cases involving the TMJ. O'SULLIVANet al. 29 reported the results of radiotherapy on 14 cases of pigmented villonodular synovitis. Their results showed that moderate dose radiotherapy (35 Gy in 15 fractions) is an effective modality in the treatment of a subset of cases with this rare condition. The most common complication after surgerY is facial paralysis. This occurred in three cases. However, facial nerve function recovery was almost complete in most cases. Although it is difficult to predict the recurrence rate of PVNS in this location because of the short follow-up periods,
our literature review shows that the recurrence rate was 9% (2 out of 22 cases). This rate is lower than that of other joints, the recurrence rates of which range from 21% 21 to 48% 35. Recurrence is presumably due to incomplete excision 19. Cases of spontaneous regression have been reported 2°. Malignant type or malignant transformation of pigmented villonodular synovitis is very rare and its existence may be debatable. BERTONI et al. 3 reported eight cases of malignant PVNS. According to them, important histologic features of malignancy were 1) a nodular, solid infiltrative pattern of the indistinct borders, 2) large, plump, round or oval cells with deep eosinophilic cytoplasm and indistinct borders, 3) large nuclei with prominent nucleoli, and 4) necrotic areas. In the region of the TMJ, one case of malignant PVNS was reported 3. This patient had four recurrences during a 7-year period. The third recurrence, deemed inoperable, was treated with 4,500 rad of radiation therapy. But the patient had pulmonary metastasis 4 years after radiation therapy. She died after the detection of metastasis, 17 years after the first diagnosis. KALIL & UNN124 reported one Case of malignant PVNS of the ankle. This patient showed lymph node metastasis. In summary, pigmented villonodular synovitis is indeed a rare lesion of the temporomandibular joint. However, we recommend that PVNS be included in the differential considerations when preauricular swelling with trismus is present. More frequent use of M R I might help diagnosis of this disease. Careful preoperative planning and complete resection are the factors that lead to the optimal treatment of PVNS.
References
1. BARNARDJDW. Pigmented villonodular synovitis in the temporomandibular joint: a case report. Br J Oral Surg 1975: 13: 183-7. 2. BEMrO~D JA, CI~ALOUPKAJC, PUT~N CM, et al. Pigmented villonodular synovitis of the temporomandibular joint: diagnostic imaging and endovascular therapeutic embolization of a rare head and neck tumor. AJNR Am J Neuroradiol 1999: 20: 159-62. 3. BERTONIF~ UNNI KK, BEABOUTJW, SIM FH. Malignant giant cell tumor of the tendon sheaths and joints (malignant pigmented villonodular synovitis). Am J Surg Pathol 1997: 21: 153-63.
P V N S in T M J 4. BREIMER CW, FREIBERGER RH. Bone lesions associated with villonodular synovitis. Am J Roentgenol 1958: 79:618 29. 5. BRAVOSM, WINALSKICS, WEISSMANBN. Pigmented villonodular synovitis. Radiol Clin North Am 1996: 34: 31156. 6. CHOONG PF, WILLEN H, NILBERT M, MERTENS F, MANDAHL N, CARLEN B, RYDHOLM A. Pigmented villonodular synovitis. Monoclonality and metastasis - a case for neoplastic origin? Acta Orthop Scand 1995: 66: 64-8. 7. CHOW LT, KUMTASM, KING WW. Extraarticular pigmented villonodular synovitis of the temporomandibular joint. J Laryngol Otol 1998: 112: 182-5. 8. CHUNG SMK, JANES JM. Diffuse pigmented villonodular synovitis of the hip joint. Review of the literature and report of four cases. J Bone Joint Surg 1965: 47A: 293 303. 9. CURTIN HI), WILLIAMSR, GALLA L, MEYERS EN. Pigmented villonodular synovitis of the temporomandibular joint. Comput Radiol 1983: 7:257 60. 10. DARLING JM, GLIMCHER LH, SHORTKROFF S, ALBANO B, GRAVALLESE EM. Expression of metalloproteinases in pigmented villonodular synovitis. Hum Pathol 1994: 25: 825-30. 11. D1NERMAN WS, MYERS EN. Pigmented villonodular tenosynovitis of the temporomandibular joint. Trans Am Acad Opthalmol Otol 1977: 84: 132-5. 12. DORWART RH, GENANT HK, JOHNSTON WH, MORRIS JM. Pigmented villonodular synovitis of synovial joints: clinical, pathologic, and radiologic features. AJR Am J Roentgenol 1984: 143: 87785. 13. DWAISKIBA S, ERIKSSON L, ELNER A, JOHANSEN CC, HANSSON LG, ~V'ESTESSON PL. Diffuse pigmented villonodnlar synovitis of the temporomandibular joint diagnosed by fine needle aspiration cytology. Diagn Cytol 1989: 5: 301-4. 14. EISlG S, DORFMAN HI), CUSAMANO RJ, KANTROWITZ AB. Pigmented villonodular synovitis of the temporomandibular joint: case report and review of the literature. Oral Surg Oral Med Oral Pathol 1992: 73: 328-33. 15. ENZINGER FM, V@ISSSW Soft tissue tumors. St Louis: C. V. Mosby, 1995:735 55. 16. FRANCHI A, FROSINI P, SANTORO R. Pigmented villonodular synovitis of the temporomandibular joint: report of a case. J Laryngol Otol 1994: 108:166 7. 17. GALLIA LJ, JOHNSON JT, MYERS EN. Pigmented villonodular synovitis of the tern-
poromandibular joint: a case report. Otolaryngol Head Neck Surg 1982: 90:691 5. 18. GEIGER S, PESCH HJ. Synovitis pigmentosa villonodularis, eine seltene Kiefergelenkerkrankung. Fortschr Kiefer Gesichtschir 1980: 25: 29-31. 19. GOLDMAN AB, DICARLO EE Pigmented villonodular synovitis: diagnosis and differential diagnosis. Radiol Clin North Am 1988: 26: 327-47. 20. GRANOWlTZ SP, D'ANTONIO J, MANKIN HL. The pathogenesis and long-term end results of pigmented villonodular synovitis. Clin Orthop 1976: 114: 335-51. 21. GREENFIELD MM, WALLACE KM. Pigmented villonodular synovitis. Radiol 1950: 54: 350-6. 22. JAFFE HL, LICHTENSTEIN L, SUTRO CJ. Pigmented villonodular synovitis, bursitis and tenosynovifis. Arch Pathol 1941: 31: 731 65. 23. JOZSA L. Immunohistochemical characterization of pigmented villonodular synovitis. Zentralbl Pathol 1992: 138: 119 23. 24. KALIL RK, UNNI KK. Malignancy in pigmented villonodular synovitis. Skeletal Radio1 1998: 27: 392-5. 25. LAPAYOWKER MS, MILLER WT, LEVY WM, HARWlCK RD. PVNS of temporomandibular joint. Radiology 1973: 108: 313 6. 26. MAKEKM, DRUMMERR. Localized nodular synovitis of the temporomandibular joint. A case report. J Maxillofac Surg 1978: 6: 302-5. 27. MIYAMOTO Y, TANI T, HAMAYA K. Pigmented villonodular synovifis of the temporomandibular joint: a case report. Hast Reconstr Surg 1977: 59: 283-6. 28. OMURAS, MIZUKI N, BUKAWAH, FUJITA K. Diffuse variant tenosynovial giant cell tumor of the temporomandibular joint: report of a case. J Oral Maxillofac Surg 1998: 56: 991-6. 29. O'SULLIVAN B, CUMMINGSB, CATTON C, BELL R, DAVIS A, FORNASIER V, GOLDBERG R. Outcome following radiation treatment for high-risk pigmented villonodular synovitis. Int J Radiat Oncol Biol Plays 1995: 32: 777-86. 30. O'SULLIVAN T J, ALPORT EC, WHISTON HG. Pigmented villonodular synovitis of the temporomandibular joint. J Otolaryngol 1984: 13: 123-6. 31. PAMMERJ, WENINGERW, HULLA H, MAZAL P, HORVAT R. Expression of regulatory apoptotic proteins in peripheral giant cell granulomas and lesions containing osteoclast-like giant cells. J Oral Pathol Med 1998: 27: 267-71.
415
32. RAIBLEY SO. Villonodular synovitis with chondromatosis. Oral Surg Oral Med Oral Pathol 1977: 44: 279-84. 33. RENAGA RUBIN I, SALAVER GIRONA A, VASQUEZ RODRIGUEZ A, ANMELLA VALMANYA J. Pigmented villonodular synovitis of the temporomandibular joint. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997: 84:459 60. 34. RICKERT RR, SHAPIRO MJ. Pigmented villonodular synovitis of the temporomandibular joint. Otolaryngol Head Neck Surg 1982: 90: 668-70. 35. SHAPIRO S, KESSLER S, MCMENOMEY SO. Pathologic quiz case 1. Temporomandibular pigmented villonodular synovitis. Arch Otolaryngol Head Neck Surgery 1996: 122: 194-96. 36. STOJADINOVIC S, RE1NERT S, WILDFORSTER U, JUNDT G, MACHTENS E. Pigmentierte villonodul/ire Synovitis des Kiefergelenks mit Einbruch in die mittlere Schiidelgrube. Mund Kiefer Gesichtschir 1998: 2:279 81. 37. SYED A, VAN HASSELT CA, TO KE Pigmented villonodular synovitis of the temporomandibular joint. J Laryngol Otol 1993: 107: 8534. 38. TAKAGI M, ISHIKAWA G. Simultaneous villonodular synovitis and synovial chondromatosis of the temporomandibular joint: report of a case. J Oral Surg 1981: 39: 699-701. 39. TANAKAK, SUZUKI M, NAMEKI H, SUGIYAMA H. Pigmented villonodular synovitis of the temporomandibular joint. Arch Otolarygol Head Neck Surg 1997: 123: 536-9. 40. YOUSSEF RE, ROSZKOWSKI M J, RICHTER KJ. Pigmented villonodular synovitis of the temporomandibuar joint. J Oral Maxillofac Surg 1996: 54: 224-7. 41. Yu GH, STAERKELGA, KERSHISNIKMM, VARMA DG. Fine-needle aspiration of pigmented villonodular synovitis of the temporomandibular joint masquerading as a primary parotid gland lesion. Diagn Cytopathol 1997: 16: 47-50.
Address: Dr. Jong-Ho Lee Department of Oral & Maxillofacial Surgery Seoul National University Hospital 28 Yeon-gun-dong, Chong-no-gu Seoul South Korea Tel." +82 2 760 3813 Fax." +82 2 766 4948 e-mail: [email protected]