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Synovial osteochondromatosis of the temporomandibular joint
212
J. Cranio-Max.-Fac. Surg. 16 (1988)
J. Cranio-Max.-Fac. Surg. 16 (1988) 212-220 © Georg Thieme Verlag Stuttgart • New York
Synovial Osteochondromatosis of the Temporomandibular Joint An Historical Review with Presentation of 3 Cases John E. de B. Norman 1, Alastair R. L Stevenson 2, Dorothy M. Painter ~, David G. Sykes 1, Leo A. Feain 1 1VMO, Mater Misericordiae (Head: Mr. J. E. de B. Norman, M. B. ChB. (Leeds), FRCS (Ed), MDS (Syd), FDSRCS (Eng), FRACDS, St. George and Royal North Shore Hospitals, and United Dental Hospital Sydney 2Department of Oral Medicine and Oral Surgery (Head: Professor M. Jolly, AM., DDSc., FRACDS), University of Sydney 3Department of Anatomical Pathology (Head: Professor A. B. P. Ng, MB, BS (Mel), FASCP, FIAC, FRCPA, Royal Prince Alfred Hospital, Sydney, Australia
Submitted 30.4. 1987; accepted 22.7. 1987
Introduction "It is very generally received opinion amongst surgeons that these substances are found in the first instance on a peduncle or footstalk, by means of which a vascular union is maintained between them and the surface of the cavities in which they are enclosed, and that it is only by the accidental rupture of this footstalk that they are set free." McCartney (1841) Scrutiny of the literature confirms that the first reference to loose bodies in the temporomandibular joint was made by Baron Albrecht (Alberto) yon Haller (1708-1777) in his magnum opus: Elementa Physiologiae corporis humani (1757-1766). Reference to this seminal work on physiology by the foremost biologist and anatomist of his century reveals the description of twenty loose bodies (glebulas) in the jaw joint of an old woman (Figs. 1 and 2). "The gland is even viscous in the shallow articular cavity of the bone of the temple, and little slime-bearing lumps are found near to the winding course of the crescent in that jointing. They are applicable to prevent the friction of parts of the body. For, on account of the everlasting movement through speaking and eating, this mode of jointing suffers intense friction, and I have observed that not only is the crescent pierced but that the incrustaceous cartilage of the temple-bone in an old woman has been completely worn away, and gathered together into almost twenty little lumps, which are held together by the membranous envelope of the jointing. The bond from the corner of the lower jaw has been driven into the muscular pillar of the tongue; it is wide, it is like skin, it is of another kind." Synovial chondromatosis is a rare cause of temporomandibular joint symptoms, and only thirty-nine cases appear to have been published in the period 1933 to the present. The first description in modern times was given by the preeminent German surgeon, George Axbausen (1933). The
Summary Loose bodies in joints have long held the fascination of surgeons and their recognition clearly enjoys a most distinguished antiquity (Pare, 1558; Hailer, !764; Barwell, 1876; Halstead, 1895) despite the rarity of their 'occurrence. This paper presents an historical review of the subject in conjunction with a report of three cases operated upon by one of the authors (JdeBN). The results have been critically reviewed by pathologists with a catholicity of experience in the field. The condition is singularly uncommon in the jaw joint, and must rank with synovial cyst and paraarticular chondroma as an unusual cause of a swelling of firstly the temporomandibular joint and secondly the parotid gland. Earlier workers recognized the value of comparative pathology to illustrate the nature of a genus of turnouts and it is educative to read the following: "A good physiological type for the loose cartilaginous bodies which infest joints is furnished by the temporomandibular joint of the skate. A recess communicating with this articular cavity usually contains a collection of smooth cartilaginous bodies, in contour and size like melon seeds." Bland-Sutton (1907) Key words Synovial chondromata - Synovial (osteo)chondromatosis - Diffuse enchondroma of joint capsule - Temporo-mandibular joint tumour - Para-articular chondroma - Loose bodies
cases published to date are listed separately within Tab. 1. Papers of general interest on the subject of osteochondromatosis include those by Fisher (1921), Mussey and Henderson (1945), Mclvor and King (1962), Murphy et al. (1962) and Mullins et al. (1965). Case Histories
Case 1. A 39-year-old woman was referred in 1975 with a history (2 years) of pain in the left temporomandibular joint. On examination, the joint was tender, swollen and crepitant. There was no limitation of mandibular movement but extreme mandibular deviation was noted. Sagittal tomography disclosed a solitary loose body in the joint and an enlarged condylar head consistent with primary hyperplasia. Straw coloured synovial fluid was aspirated at operation and a condylectomy and arthroplasty were performed with removal of a very large solitary loose body. This had a concavo-convex configuration conforming with the caput mandibulae and glenoid fossa respectively. No temporomandibular meniscus was demonstrated during the operation. Histological examination confirmed active condylar hyperplasia and synovial osteochondromatosis. A portion of the loose body had become ossified.
Case2. A 39-year-old woman was referred in 1978 with a history (1 year) of pain in the left temporomandibular
Synovial Osteochondromatosis of the TemporomandibuIar Joint
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PHYSIOLOGI.,E CORPORIS
J. Cranio-Max.-Fac. Surg. 16 (1988)
T U S.
D E G L U T I T I O . VEN3'R1CI~LUS. OMENTA, LIEN. PANCREAS. HEPA, R.
8EOZ L MANDUCAT~O.' quent~r Inl'pe@~aaparent, veri~ cttm carttl~ginibtt~lategr, rtperl. Sed ctiam qua contra R t v z u ~ fentiunt, illui~rtm virl, tea tamen rein h~. terpretantur, ut & cure finu,& cam tubercalo muiilam a~ant dear. ac~lati ( 1 ), S. V I I.
Llgamtnra eju~ artic~t~rtionlt.
Prater ~pfulam attlcdh omnibu~ eommuaem, el~ in maxilla~ tnferiori~ articulo ligaraentum lnfigne, laterale ( m ) , ortum ab offe temporura, ¢xqu¢ ejus fine, quem dixtmus, peRerius & inferius, quam articulatto iiafefioria maxilla, & infettum in condyli maxillz i~fferiori= margiaem pofleti0rem, inter angalam & condylum, lepta mufcalum pterygo !deam intemum. S~per hoe ligamentum atteria maxillaris iaterna fubit'. Duo llgImenta alia defcriblt CI, F~ ~ a z t s ( n ), nova & peealiaria, rata ab eminenfla tranav¢ffall, cui maxilla adaptatur, interiu= aliud, elterum exterttts, & tnferta in extremum ¢ondylum. Ea ~identat alia CL yiri cam capfuls numeraffe. Glandula eti~m mncilaglnofa in g|ena ( a ) oflh temporum, glebul= juxta menffci ambimat muciferz ( t ) in ea articuhtione rq~. Iriunmr, Pertinent ad friS:ionem impediendam. Ob perpemum enim in Io; quendo & edendo mature, h~c ardculatio magnam adtridonem patimr, neque (alum menifcum perforatom, fed omnino crab.am cartilagineam Orris temporum in vetula detrlt~m vidl, inque viginti fete g!ebulaa col. 'ld.hm, qu~ c~pfula artic~li comprehendebantur. Lig.~menmm ab zngulo maxiilz tnteriorls i~ n:,fculnm flytog!offam demi,~m ( r ) , htum~ membra~a~m, alterius nat~r~ eft.
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Fig. 1 Title page from Elementa Physiologiae Corporus Humani. Sumptibus Societatis Typographicae, Bernae 1764, Alberto von Hailer published by courtesy of the Librarian of The Royal Society of Medicine (Photographic Unit RSM).
Fig.2 von Halter's reference to loose bodies in the temporomandibular joint. (See text for translation)•
joint. On examination, the left joint was swollen, tender and crepitant. There was no limitation of mandibular movement. Radiographic examination confirmed osteoarthritic change, but not discernible loose bodies. Straw coloured synovial fluid was aspirated at operation and an arthrotomy disclosed eight small and irregular loose bodies. Recovery was uneventful and histological examination confirmed synovial chondromatosis.
joint, the left acromioclavicular joint and the left coracoid process (Fig. 8). An arthrotomy was performed and fiftytwo loose bodies removed (Fig. 9). The wound was drained and closed in layers. The patient was discharged on the 4th day operatively. The tissue was examined by three consultant pathologists and the following composite report has been given: Macroscopic appearance: The specimen consists of loose bodies from the left temporomandibular joint. There were 52 pieces of pale cartilaginous tissue weighing in aggregate 3.4 grams and varying in size from 3-15 mm. in maximum diameter and showing a smooth cobblestone surface. Microscopic appearance: The sections showed nodules of cartilaginous tissue, some with fibrinous surface tags, and two with quite extensive cystic degenerative change. Each had a squamoid epithelial surface. The cartilage cells were pleomorphic and many individual cells showed mineralization. No bizarre cells were present and mitoses were not seen. The central portions of the larger nodules had early mineralization of matrix as well as cartilage cells, but true bone formation was not present in these fragments.
Case 3. A 59-year-old woman was referred with a history of increased pain and swelling in the left temporomandibular joint resulting in mandibular deviation to the right and a left posterior open bite. The symptoms had developed over the preceding six months following a heavy fall in which she fractured her clavicle and damaged the left jaw joint. On examination there was an obvious preauricular swelling, 3 cm. in diameter, situated over the joint (Fig. 7). Tomographic examination confirmed both osteoarthritic changes and the presence of multiple radiopaque loose bodies. A bone study with single photon emission computed tomography (SPECT) was carried out with 800 MBq of technetium 99 m MDP. This bone scan was strongly positive for the affected temporomandibular
214
J. Cranio-Max.-Fac. Surg. 16 (1988)
Table 1
S u m m a r y of 42 c a s e s ( 1 7 6 4 - 1 9 8 7 ) w i t h t a b u l a t i o n of s a l i e n t p o i n t s
Paper
No.
Year
Sex Age Side Length of Synovial history in fluid years
Hailer 1 Axhausen 2
1764 1933
F M
21
R
Trevor Schneider ,~chneider Feist
3 4
1952 1960
F F
63 52
R R
4/12
5
1960
F
47
R
6
1960
F
59
Inovay
7
1962
F
Schulte
8
1969
F
J.E. de B. Norman et al.
Pain
Swelling
+
+
+
+
+
+
3
+
+
R
1
+
+
46
L
7/12
-
+
31
R
5
+
--
Crepitus
1 3
12 13
1972 1973
F F
37 53
L R
2/12 19
+
+ -
+
Tasanen 14
1974
F
40
R
5
+
-
+
2
Lomba
17
1977
F
32
R
1
Akhtar
18
1977
F
53
R
2
Ronald Ronald Ronald
19 20 21
1978 1978 1978
M M M
49 47 75
R
2 3/12 10/12
Thick clear amber Three aspirations
+
+
-
+
+
÷
÷
+
÷
+
+
A+C÷ Gelfoam
+ +
Ballard Ailing
L
Silastic A
1
4/12
62
A+C+
-
R
F
12x
4--.14
42
1977
1
7
M
16
+
+
1972
Raibley
+
Repeated dislocation, deviation Inability to close, deviation Deviation -
11
+
+
-
Ballard
cms) +
40~80
480
1
+
-
-
R
4/12
A+S
-
-
18
-
(2,5
A
-
-
M
L
10x 10x15 25x15
+
1971
44
1
+ Deviation +
-
+
1
+
+
(Concretion) 15
+
--
+
+
No histology No histology
2 Bodies le~ in situ Diagnosis benign synovialoma
A
3x2x 2~13 xl0x 3 25x10 x I0
A+C+S A
A
1)A 2)A+C Resection of tumour
29
4
Special features
A+C+M
8x5 0.5~4
10
F
+
-
Silver
1977
A+M+S
-
5
15
80
-
R
Rosen
-
+
-
28
+ (2.5 cms)
-
+
+
F
+
Multiple
+
1969
+ (2cms) -
A+S
+
-
9
+
-
-
+
Operations
20 Numerous 48
+
Kusen
Clear (8mls)
Limitation of Radiology Number Size manOsteo Loose loose mm. dibular arthritis bodies bodies movement
Diagnosis villonodular synovitis
A+S
5×5x 3410 x8 x5
1)SP 2)A
A+C A+C +
+
+
+
+
10
3~ lx17
+
20~30
245
A+C+M+ S+ Silastic A
+
25
1~5
A
+
1
+
1
Deviation
Ronald Ronald
Miller
22 23
1978 1978
F F
32 48
R
4/12 2
Cloudy (1 ml.)
+ +
+ +
+
Deviation
24
1978
F
61
L
10
Blenkin- 25 sopp
1978
M
60
L
1/12
Olley
26
1978
F
20
R
3/12
Fee
27
1979
F
48
R
1
÷
÷
Galliard 28 Takagi 29
1981 1981
F M
66 36
R R
6 3
--
+
+
+
+
+
Straw coloured Greenish gelati-
+ + (3 cms) ÷ + (3 cms)
--
--
+
nous
Deviation
-
-
+
(4x4 cms) Brants Morrish
36 31
1982 1983
M F
42 47
L L
25 6/12
+
Kraszewski
32
1984
F
49
R
6
÷
+
+
+
Deviation
--
÷
+ Deviation
+ (Scan +)
+
A
A+C+S+ Silastic 50 2~5 A 1)A+C Diagnosis(partial) villonodular 2 ) A + C synovitis (total) 70 A+S Several 3 ~ 1 5 A+C+ Gelfoam 107
20
A
Diagnosis osteochondritis dessicans
8ynovial Osteochondromatosis of the Temporomandibular Joint Table I
J. Cranio-Max.-Fac. Surg. 16 (1988)
215
Continued
Paper
No.
Year
Sex Age Side Length of Synovial Pain history in fluid years
Swelling
Crepitus
Limitation of Radiology Number Size manOsteo Loose loose ram. dibular arthritis bodies bodies movement
Operations
Kraszewski Blankestijn Btankestijn Blankestijn Oe Bont
33
1984
M
40
L
4
-
-
+
Deviation
A+M
34
1985
M
59
L
3/12
-
+
.
35
1985
M
19
R
18/12
+
+
-
36
1985
M
39
L
7
-
+
+
37
1985
F
63
R
4
Mucus
.
.
.
.
.
Deviation .
+
(Scan +) -
+ -
.
1+
40x30 x 30
20
1 ) A + SP
3
2) A
Numerous Multiple
A+C+S
Special features
A+C+ S
10
143.5
A+C+S
100
2x2x 542 x 2x8 1)SP 2)A+ S+M+ Silastic
A+M
U.C.H.
(Scan
+) Silver
38
1986
F
38
L
8/12
+
-
+
+
-
-
Thompson
39
1986
M
59
L
3
-
+ (2.5 x 3cms)
-
-
(Scan +)
Multiple2x3
Norman 40
1987
F
39
L
2
+
+
-
-
+
1
Norman
41
1987
F
39
L
1
+
+
-
+
-
8
9
Norman
42
1987
F
59
L
6/12
Straw + Coloured Straw + Coloured Straw + Coloured
+
52 (3.4g)
A+S
A C M S SP UCH
Arthrotomy Condylectomy Meniscectomy Synovectomy Superficial parotidectomy Unilateral condylar hyperplasia
= = = ~ ~ =
Scan + Blank
+ + (3cms)
= = = =
Left + Unilateral (Scan Open Bite, + ) Right Deviation
Technetium 99 m bone scan Present Not present Not mentioned
The periphery of the nodules was composed of fibrocartilage and one of the nodules had a peripheral focus of vascularity and cellularity which resembled granulation tissue (pannus formation). Diagnosis: temporomandibular joint - synovial (osteo) chondromatosis. Discussion Synovial choudromatosis may present with articular pain and preauricular swelling (mimicking a parotid tumour), crepitus and limitation of movement, with deviation to either side productive of a subtle degree of laterognathism. The swelling may be hard or fluctuant, and will probably move with the condyle or even disappear momentarily with mouth opening. A solitary loose body is rarely palpable, contrary to experience with larger joints. The mass does not transilluminate, and there is no associated muscle atrophy. Plain films and tomography of the joint (Figs. 3 and 4) may demonstrate the loose bodies, one or many (up to 480 have been described); and scintigraphy will usually show an area of increased uptake as illustrated in the accompanying photographs (Figs. 5 and 6). The use of CT scanning and in cases of diagnostic dubiety CT sialography is not disputed, and will clearly be of immeasurable value in cases where the mass is thought to arise from the parotid gland. Preliminary diagnostic arthroscopy would seem to be unnecessary in the presence of a good Mstory, physical and radiographic findings.
DiagnosisMixed Parotid Tumour C+S
U.C.H.
A+8
summary female patients male patients right side left side side not stated
28 14 22 16 4
Joint Fluid There may be an obvious effusion, although the state of the synovial fluid has not been the object of comment in most of the reported cases. In our three examples the synovial fluid was straw coloured, and in each instance it was of increased volume and viscosity, albeit not fibrin flecked. In the third case, there were a few pus cells present and the Rheumaton test of the fluid was negative.
Nodules The nodules may be pearly white, grey, or of a pink or light azure hue. They are of variegated shape and ranging in size from 1 mm. to 1.5 cm. or larger, and in the latter case may conform to the configuration of the caput, the fossa, or both. The meniscus or disc may be absent in such instances and when present in our cases it was not perforated. The synovium is generally thickened and the glenoid fossa and articular eminence denuded or even studded with diminutive nodules in various stages of development, including some of petiolar form. This was observed in the most recent arthrotomy of the series and the embryonic nodules were, for the most part, readily detached from the vault with a Volkmann's spoon. Conglomerate mass lesions of osteocartilaginous material were not encountered in this series, although they have been described. Multiple synovial chondromata may be found in tendon sheaths, bursae and at a distance from the joint (Geshickter and Copeland, 1949; Lichtenstein, 1972). Only one para-articular chondroma arising from the tern-
216
J. Cranio-Max.-Fac. Surg. 16 (1988)
Fig. 3 Sagittal tomogram of the left temporomandibular joint showing enlargement of the joint space, forward positioning of the caput in addition to an aggregate of loose bodies,
Fig.5 Single photon emission computed tomography (SPECT) of skull and jaw with 800 MBq Tc99 m MDP which is strongly positive for the left temporomandibularjoint (image 12).
J.E. de B. Norman et al.
Fig.4 Coronal tomogram of the same joint showing lamellar structure of loose body.
Fig.6 Radionuclide scan (SPECT) showing identical findings (image22).
poromandibular joint, and invading the parotid gland, has been seen in over three hundred operations on the jaw joint and a further combined series of two hundred and fifty turnouts of the major and minor salivary glands (Norman and Williams, 1984). The same series includes a solitary synovial cyst arising from the temporomandibular joint and invading the parotid, presenting as a salivary gland mass and requiring superficial parotidectomy and facial nerve dissection. Diagnostic confusion may occur, and it is educative to review the thoughtful paper by Thompson et al. (1986) in which an intra parotid mass was first thought to be a pleomorphic salivary adenoma and later an example of synov-
Fig.7 Photograph of an obvious preauricular swelling.
ial chondromatosis. We suggest that the initial lesion had the characteristic appearance of a para-articular chondroma and was remarkably similar to a case in our own series.
Pathogenesis of Loose Bodies Synovial chondromatosis has been described in primary and secondary forms. Primary the synovium develops from the embryonic mesenchyme and as a consequence of metaplasia, cartilaginous foci develop within the synovial membrane. An intra-synovial spherical body slowly forms with the villous process of the synovial membrane as its pedicle. This later becomes
Synovial Osteochondromatosisof the TemporomandibularJoint
J. Cranio-Max.-Fac.Surg. 16 (1988)
217
Fig.8 Radionuclide scan (SPECT) of same patient strongly positive for left acromioclavicular joint and left coracoid process (recent trauma).
Fig. 9 Intraoperative photograph showing extent of the arthrotomy and accumulation of multiple loose bodies displayed on an huckaback towel,
detached and falls into the joint cavity where it becomes enveloped by a perichondrium and is nourished by diffusion of the synovial fluid. The chondroblasts and osteoblasts proliferate and enlargement occurs by surface layering. Resorption can occur as the result of osteoclastic activity and may produce surface irregularity. Necrosis of the central area may occur as the result of deprivation of synovial nutrition. Although calcification may follow in this zone, re-attachment and re-vascularization have been described. Dahlin (19 81) reports that "histologic appearance of these proliferating tumours is what makes them important to the pathologist. In many instances the nuclear abnormalities and numbers of multinucleated cells are so great that in the right context, as in the medulla of a major tubular bone, the diagnosis of chondrosarcoma would be strongly supported. Paradoxically, however, these cartilaginous masses are practically never malignant nor even premalignant." This is an active metaplastic rather than neoplastic process without apparent underlying disease. Milgram (1977 a, b, c) recognizes three phases: (a) Active synovial disease without loose bodies. (b) Synovial nodules with loose bodies. (c) Multiple loose bodies with inactive synovial disease. The number of loose bodies varies and the record of 1047 in a single joint appeared to be held by Berry (1891, 1894) until we came upon Bland-Sutton's report (1907) of 1532 loose bodies in a shoulder joint. Our modest 52 loose bodies pales into insignificance. Bilaterality is rare, albeit described by Humpbry (1888) in relation to the elbow joint. The first unilateral case described in the Australian literature may have been by Syme (1888). It rarely occurs in children and the youngest patients in
those cases involving large joints were 5 and 8 years respectively. Secondary synoviaI chondromatosis is a more passive process involving synovial nourishment of dislodged fragments of joint tissue. The latter may be secondary to trauma or to joint disease such as osteochondritis dessicans and inflammatory or non inflammatory arthropathy. The dislodged bone or cartilage fragments undergo concentric layering. They may lodge in the synovium with consequent cartilaginous metaplasia and nodule formation which is more irregular than that of primary chondromatosis and shows no cytological atypia.
Treatment "The treatment is essentially surgical. In the literature we have noted such radical procedures as amputation, resection and synovectomy. We have removed the bodies by the simplest possible route, and as recurrences have been so rare we see no need to change our procedure." Henderson and Jones (1923) The observation made by two orthopaedic surgeons from the Mayo Clinic 64 years ago in relation to procedures upon large weight bearing joints remains apposite (Jones, 1924). The management of the condition is surgical and whilst we have no doubt that removal of loose bodies from the jaw joint via the operating arthroscope may be the technique of the future, the delicate nature of the needle arthroscopes presently in use precludes all but the smallest joint mice being retrieved per arthroscopy. Arthrotomy, therefore, is indicated and the joint exposed via a standard temporofacial incision as described elsewhere (AI-Kayat and Bramley, 1979; Norman, 1982). Comfortable surgical access and working space is essential as the furthest reaches of the joint need to be examined
218
J. Cranio-Max.-Fac. Surg. 16 (1988)
Fig. 10 Photomicrograph of loose body (case 3) with dense cartilage and an early mineralized margin contained within synovium. (HE x20).
Fig, 12 Photomicrograph of cartilage island surrounded by fibrin and embedded in synovium. Some of the cartilage cells are pleomorphic and the stroma is very slightly mineralized (per kind favour of Mr. A. Shehata and Dr. R. Holt). (HE ×40). '
and any occult loose body removed. The authors favour the temporofacial incision, with splitting of the temporalis muscle to allow relaxed forward dissection along the zygomatic arch at the subperiosteal level. The glenoid lobe of the parotid gland and the contained temporofacial ramus of the seventh cranial nerve are swept forward from the joint capsule with a Dickson-Wright dissector. Where an effusion is encountered or loose bodies are anticipated, an aspiration is carried out prior to arthrotomy and the synovial fluid submitted for examination. Upon incising the joint capsule, the surgeon will almost certainly be rewarded by a flow of viscid synovial fluid followed by the egress of many pearly white bodies, some smooth, some slightly faceted and some bossellated. A thorough examination of the joint will require forward displacement of the condyle (by the assistant) in order that every nook and cranny be inspected. Movement of the condyle and gentle massaging of the overlying tissues will produce further bodies and this condylar movement and massage is considered desirable in order that any material
J.E. de B. Norman et al.
F i g . l l Multiple cartilage islands, some with pleomorphic cells, embedded in synovium (per kind favour of Mr. A. Shehata and Dr. R. Holt). (HE x 20).
still adherent by a footstalk will be detached. The joint is repeatedly irrigated and inspected. The surgeon should not refrain from a limited synovectomy and must carefully inspect the vault. In the third patient we noted numerous embryonic bodies in this area, a few of which were attached by a pedicle, and this zone was scarified with a Volkrnann's spoon. It is unnecessary to remove the mandibular condyle for this benign condition, although a limited synovectomy is advisable. We consider that a total synovectomy is impossible of achievement with the mandibular condyle in situ, and there is no valid indication to remove it. The authors recommend a partial or limited synovectomy and the accessible synovial lining is dissected out with fine micro scissors (Owen-Vickers type) and bleeding points are sealed with the bipolar diathermy. The procedure is carried out under magnification. At the conclusion of the operation, the surgeon may wish to inspect the furthest reaches of the joint with the arthroscope and we have employed the Wolf Lumina SL 1.9 ram. telescope over the triennium for diagnostic arthroscopy. Following meticulous haemostasis, the wound is carefully closed in layers with suction drainage. A temporo-mastoid dressing is applied and the patient discharged on the 4th post-operative day to be followed up at the out-patient clinic. A puree diet is advised for the first week and normal sustenance thereafter.
Conclusions An historical review of synovial osteochondromatosis affecting the temporomandibular joints has been presented with a review of the relevant literature. Although the condition is of singular rarity, the physical signs are relatively characteristic and include pain, swelling and crepitus. Plain films may not adequately demonstrate the loose bodies and the clinician may need to resort to sagittal tomography or even CT scanning of the joint. The treatment of synovial chondromatosis is surgical and the loose bodies should be removed by arthrotomy with meticulous examination of the joint cavity. We consider it unnecessary to
Synovial Osteochondromatosis o f the Temporomandibular Joint remove the m a n d i b u l a r condyle for this benign condition, although a limited synovectomy m a y be advisable. Acknowledgements Appreciation and thanks are due to Professor B.E.D.Cooke, FRCPath., for advice in the preparation of this paper. The photomicrographs illustrating an embryonic form of osteochondromatosis from the finger joint are published by kind permission of Dr, R.S. Hoh, DCP (Lond.), and Mr. N.G.Shehata, FRCS., FRACS, Thanks are due to Mrs. Elizabeth de Kantzow, BA (Syd.), Grad. Dip. Lib. Sc., ALAA., for constructive criticism in the preparation of the text and bibliography. The scholarship of Ronald A. Elliott, Esq., BA (Syd.), in translating the Latin text is acknowledged with pleasure. The photographs have been taken by Mr. Timothy CoIlis-Bird, and the manuscript typed by Mrs. Jocelyn Hungerford and Miss Gabriella Kalmar.
References
Akbtar, M., S. Mahajan, E. Kott: Synovial chondromatosis of the temporomandibular joint. J. Bone Joint Surg. (Am.) 59-A (1977) 266 AI-Kayat, A., P. Bramley: A modified pre~auricular approach to the temporomandibular joint and malar arch. Br. J. Oral Surg. 17 (1979) 91 Ailing, C. C., D. W. Rawson, O. J. Staats, 1L A. Middleton: Clinicalpathological conference, case 2. J. Oral Surg. 31 (1973) 604, 691 Axhausen, G.: Pathologie und Therapie des Kiefergelenkes. Fortschr. Zahnheilk. 9 (1933) 171 Ballard, R., L. H. Weiland: Synovial chondromatosis of the temporomandibular joint. Cancer 30 (1972) 791 Barwell, R.: Movable bodies in joints. Br. Med. J. 1 (1876) 184 Berry, J.: Fifty loose bodies removed from a knee-joint. Tr. Path. Soc. London 42 (1891) 275 Berry, J.: Multiple loose bodies from the knee-joint. Br. Med. J. 1 (1894) 1081 Bland-Sutton, J.: Connective tissue tumours - chondromata. In: Tumours innocent and malignant 4th Ed. Cassell, London (1907) 33 Blankestijn, J., A. K. Panders, A. Vermey, A. J. J. A. Scherpbier: Synovial chondromatosis of the temporo-mandibular joint: Report of three cases and a review of the literature. Cancer 55 (1985) 479 Blenkinsopp, P. T.: Loose bodies of the temporo-mandibular joint, synovial chondromatosis or osteoarthritis. Br. J. Oral Surg. 16 (1978) 12 Brants, J. F., G. J. Kusen: Synoviale chondromatosis van het kaakgewricht. Acta Stomatol. Belg. 79 (1982) 37 Dahlin, D. C.: Conditions that simulate primary neoplasms of bone. In: Bone Tumors 3rd. Ed. Thomas., Springfield (1981) 405 De Bont, L. G. M., J. Blankestijn, A. K. Panders, A. Vermey: Unilateral condylar hyperplasia combined with synovial chondromatosis of the temporomandibular joint: Report of a case. J. Maxillofac. Surg. 13 (1985) 32 Fee, W. E., P. Windhorst, R. Wiggins, L. Pang: Synovial chondromatosis of the temporomandibular joint. Otolaryngol. Head Neck Surg. 87 (1979) 741 Feist, J. H., T. G. Gibbons: Osteochondromatosis of the temporomandibular joint. Radiology 74 (1960) 291 Fisher, A. G. T.: A study of loose bodies composed of cartilage or of cartilage and bone occurring in joints, with special reference to their pathology and aetiology. Br. J. Surg. 8 (1921) 493 Gaillard, A., J. Delaire, D. Jacquemaire, J. M. Buzelin: La chondromatose synoviale de l'articulation temporo-mandibulaire. Rev. Stomatol. Chir. Maxillofac. 82 (1981) 105 Geshickter, C. G., M. M. Copeland: Tumors of tendon sheaths, joints and bursae. In: Tumors of Bone 3rd Ed. Lippincott, Phildadelphia (1949) 697
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Hailer, A. yon: Elementa Physiologiae Corporus Humani. Sumptibus Societatis Typographicae, Bernae (1764) 8 Halstead, A. E.: Floating bodies in joints. Ann. Surg. 22 (1895) 327 Henderson, M. S., H. T. Jones: Loose bodies in joints and bursae due to synovial osteocbondromatosis. J. Bone Joint Surg. 21 (1923) 400 Humphry, G. M.: Loose bodies in joints. Br. Med. J. 2 (1888) 707 Inovay, J.: A rare case of benign synovialoma of the mandibular joint. Oral Surg. 15 (1962) 775 Jones, H. T,: Loose body formation in synovial osteochondromatosis with special reference to the etiology and pathology. J. Bone Joint Surg. 22 (1924) 407 Kraszewski, J., G. Krzymanski, G. Lomzynski, J. Walecki: Chrzesmiakowatosc maziowki stawu skroniowo-zuchwowego. Czas. Stomatol. 37 (1984) 349 Kusen, G. J.: Chrondromatosis: Report of case. J. Oral Surg. 27 (1969) 735 Lichtenstein, L.: Tumors of synovial joints, bursae and tendon sheaths. In: Bone Tumors 4th Ed. Mosby., St. Louis (1972) 409 Lomba, J. A., R. Cabanas, L Marrero: Synovial chondrometaplasia of the temporomandibular joint: Report of a case. J. Oral Surg. 35 (1977) 675 McCartney, J.: On the movable cartilaginous bodies found in the synovial and serous cavities. Tr. Prov. Med. and Surg. Assn. 9 (1841) 427 Mclvor, R. B., D. King: Osteochondromatosis of the hip joint, J. Bone Joint Surg. (Am.) 44-A (1962) 87 Milgram, J. W.: The classification of loose bodies in human joints. Clin. Orthop. 124 (1977 a) 282 Milgram, J. W.: The development of loose bodies in human joints. Clin. Orthop. 124 (1977 b) 292 Milgram, J. W.: Synovial osteochondromatosis: A histopathological study of thirty cases. J. Bone Joint Surg. (Am.) 59-A (1977 c) 792 Miller, A. S., R. D. Harwick, D. J. Daley: Temporomandibular joint synovial chondromatosis: Report of a case. J. Oral Surg. 36 (1978) 467 Morrish, R. B., L. S. Flamen, W. H. Ware: Synovial chondromatosis of the temporomandibular joint. J. Craniomandibular Pract. 2 (1983) 64 MuIlins, F., C. W. Berard, S. H. Eisenberg: Chondrosarcoma following synovial chondromatosis. Cancer 18 (1965) 1180 Murphy, F. P., D. C. Dahlin, C. R. Sullivan: Articular synovial chondromatosis. J. Bone Joint Surg. (Am.) 44-A (1962) 77 Mussey, R. D., M. S. Henderson: Osteochondromatosis. J. Bone Joint Surg. 31-A (1949) 619 Norman, J. E. deB.: Post-traumatic disorders of the jaw joint. Ann. R. Coll. Surg. Eng. 64 (1982) 29 Norman, ]. E. de B., 1L G. Williams: Tumours of the major and minor salivary and lacrimal glands - a two centre survey of 250 cases over the period 1962-1984. Abstr. European Association for Ma×illo-Facial Surgery, 7th Congress September 1984, Paris Olley, S. F., P. J. Leopard: Osteochondritis dessicans affecting the temporo-mandibular joint. Br. J. Oral Surg. 16 (1978) 21 ParS, A.: (1558) (quoted by Barwell). Raibley, S. O.: Villonodular synovitis with synovial chondromatosis. Oral. Surg. 44 (1977) 279 Ronald, J. B., E. E. Keller, L. H. Weiland: Synovial chondromatosis of the temporomandibular joint. J. Oral Surg. 36 (1978) 13 Rosen, P. S., K. P. H. Pritzker, J. Greenbaum, R. C. Holgate, A. M. Noyek: Synovial chondromatosis affecting the temporomandibular joint. Arthritis Rheum. 20 (1977) 736 Schneider, G.: Zur Chondromatose des Kiefergelenks. Dtsch. Zahnfirztl. Z. 15 (1960) 1233 Schuhe, W. C., tL R. Rhyne: Synovial chondromatosis of temporomandibular joint. Oral. Surg. 28 (1969) 906 Silver, C. M., S. D. Simon, H. M. Litehman, J. Dyckman: Synovial chondromatosis of the temporomandibular joint. J. Bone Joint Surg. (Am.) 53-A (1971) 777 Silver, C. M., M. Motamed, D. E. Moonan: Chondromatosis of the temporomandibular joint arising in the meniscus. J. Oral Ma×illofac. Surg. 44 (1986) 70
220
y. Cranio-Max.-Fac.
Surg. 16 (1988)
Syme, G. A.: A case of loose body removed from the elbow-joint, presumably due to fracture of the head of the radius. Aust. Med. J. 10 (1888) 323 Takagi, M., G. Ishihawa: Simultaneous villonodular synovitis and synovial chondromatosis of the temporomandibular joint: Report of case. J. Oral Surg. 39 (1981) 699 Tasanen, A., M. A. Lamberg, R. Kotilainen: Osteochondromatosis of the temporomandibular joint. Oral Surg. 38 (1974) 845 Thompson, K., H. C. Schwartz, J. W. Miles: Synovial chondromatosis of the temporomandibular joint presenting as a parotid mass:
J. E. de B. N o r m a n et al.: Synovial Osteochondromatosis possibility of confusion with benign mixed tumor. Oral Surg. 62 (1986) 377 Trevor, D.: A case of synovial chondromatosis of the temporomandibular joint. Postgrad Med. J. 28 (1952) 408
Mr. J. E. de B. Norman, FRCS (Ed.) 9 West Street, Hurstville New South Wales, 2220 Australia
J. Cranio-Max.-Fac. Surg. 16 (1988)
J. Cranio-Max.-Fac. Surg. 16 (1988) 212-220 © Georg Thieme Verlag Stuttgart • New York
Synovial Osteochondromatosis of the Temporomandibular Joint An Historical Review with Presentation of 3 Cases John E. de B. Norman 1, Alastair R. L Stevenson 2, Dorothy M. Painter ~, David G. Sykes 1, Leo A. Feain 1 1VMO, Mater Misericordiae (Head: Mr. J. E. de B. Norman, M. B. ChB. (Leeds), FRCS (Ed), MDS (Syd), FDSRCS (Eng), FRACDS, St. George and Royal North Shore Hospitals, and United Dental Hospital Sydney 2Department of Oral Medicine and Oral Surgery (Head: Professor M. Jolly, AM., DDSc., FRACDS), University of Sydney 3Department of Anatomical Pathology (Head: Professor A. B. P. Ng, MB, BS (Mel), FASCP, FIAC, FRCPA, Royal Prince Alfred Hospital, Sydney, Australia
Submitted 30.4. 1987; accepted 22.7. 1987
Introduction "It is very generally received opinion amongst surgeons that these substances are found in the first instance on a peduncle or footstalk, by means of which a vascular union is maintained between them and the surface of the cavities in which they are enclosed, and that it is only by the accidental rupture of this footstalk that they are set free." McCartney (1841) Scrutiny of the literature confirms that the first reference to loose bodies in the temporomandibular joint was made by Baron Albrecht (Alberto) yon Haller (1708-1777) in his magnum opus: Elementa Physiologiae corporis humani (1757-1766). Reference to this seminal work on physiology by the foremost biologist and anatomist of his century reveals the description of twenty loose bodies (glebulas) in the jaw joint of an old woman (Figs. 1 and 2). "The gland is even viscous in the shallow articular cavity of the bone of the temple, and little slime-bearing lumps are found near to the winding course of the crescent in that jointing. They are applicable to prevent the friction of parts of the body. For, on account of the everlasting movement through speaking and eating, this mode of jointing suffers intense friction, and I have observed that not only is the crescent pierced but that the incrustaceous cartilage of the temple-bone in an old woman has been completely worn away, and gathered together into almost twenty little lumps, which are held together by the membranous envelope of the jointing. The bond from the corner of the lower jaw has been driven into the muscular pillar of the tongue; it is wide, it is like skin, it is of another kind." Synovial chondromatosis is a rare cause of temporomandibular joint symptoms, and only thirty-nine cases appear to have been published in the period 1933 to the present. The first description in modern times was given by the preeminent German surgeon, George Axbausen (1933). The
Summary Loose bodies in joints have long held the fascination of surgeons and their recognition clearly enjoys a most distinguished antiquity (Pare, 1558; Hailer, !764; Barwell, 1876; Halstead, 1895) despite the rarity of their 'occurrence. This paper presents an historical review of the subject in conjunction with a report of three cases operated upon by one of the authors (JdeBN). The results have been critically reviewed by pathologists with a catholicity of experience in the field. The condition is singularly uncommon in the jaw joint, and must rank with synovial cyst and paraarticular chondroma as an unusual cause of a swelling of firstly the temporomandibular joint and secondly the parotid gland. Earlier workers recognized the value of comparative pathology to illustrate the nature of a genus of turnouts and it is educative to read the following: "A good physiological type for the loose cartilaginous bodies which infest joints is furnished by the temporomandibular joint of the skate. A recess communicating with this articular cavity usually contains a collection of smooth cartilaginous bodies, in contour and size like melon seeds." Bland-Sutton (1907) Key words Synovial chondromata - Synovial (osteo)chondromatosis - Diffuse enchondroma of joint capsule - Temporo-mandibular joint tumour - Para-articular chondroma - Loose bodies
cases published to date are listed separately within Tab. 1. Papers of general interest on the subject of osteochondromatosis include those by Fisher (1921), Mussey and Henderson (1945), Mclvor and King (1962), Murphy et al. (1962) and Mullins et al. (1965). Case Histories
Case 1. A 39-year-old woman was referred in 1975 with a history (2 years) of pain in the left temporomandibular joint. On examination, the joint was tender, swollen and crepitant. There was no limitation of mandibular movement but extreme mandibular deviation was noted. Sagittal tomography disclosed a solitary loose body in the joint and an enlarged condylar head consistent with primary hyperplasia. Straw coloured synovial fluid was aspirated at operation and a condylectomy and arthroplasty were performed with removal of a very large solitary loose body. This had a concavo-convex configuration conforming with the caput mandibulae and glenoid fossa respectively. No temporomandibular meniscus was demonstrated during the operation. Histological examination confirmed active condylar hyperplasia and synovial osteochondromatosis. A portion of the loose body had become ossified.
Case2. A 39-year-old woman was referred in 1978 with a history (1 year) of pain in the left temporomandibular
Synovial Osteochondromatosis of the TemporomandibuIar Joint
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213
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PHYSIOLOGI.,E CORPORIS
J. Cranio-Max.-Fac. Surg. 16 (1988)
T U S.
D E G L U T I T I O . VEN3'R1CI~LUS. OMENTA, LIEN. PANCREAS. HEPA, R.
8EOZ L MANDUCAT~O.' quent~r Inl'pe@~aaparent, veri~ cttm carttl~ginibtt~lategr, rtperl. Sed ctiam qua contra R t v z u ~ fentiunt, illui~rtm virl, tea tamen rein h~. terpretantur, ut & cure finu,& cam tubercalo muiilam a~ant dear. ac~lati ( 1 ), S. V I I.
Llgamtnra eju~ artic~t~rtionlt.
Prater ~pfulam attlcdh omnibu~ eommuaem, el~ in maxilla~ tnferiori~ articulo ligaraentum lnfigne, laterale ( m ) , ortum ab offe temporura, ¢xqu¢ ejus fine, quem dixtmus, peRerius & inferius, quam articulatto iiafefioria maxilla, & infettum in condyli maxillz i~fferiori= margiaem pofleti0rem, inter angalam & condylum, lepta mufcalum pterygo !deam intemum. S~per hoe ligamentum atteria maxillaris iaterna fubit'. Duo llgImenta alia defcriblt CI, F~ ~ a z t s ( n ), nova & peealiaria, rata ab eminenfla tranav¢ffall, cui maxilla adaptatur, interiu= aliud, elterum exterttts, & tnferta in extremum ¢ondylum. Ea ~identat alia CL yiri cam capfuls numeraffe. Glandula eti~m mncilaglnofa in g|ena ( a ) oflh temporum, glebul= juxta menffci ambimat muciferz ( t ) in ea articuhtione rq~. Iriunmr, Pertinent ad friS:ionem impediendam. Ob perpemum enim in Io; quendo & edendo mature, h~c ardculatio magnam adtridonem patimr, neque (alum menifcum perforatom, fed omnino crab.am cartilagineam Orris temporum in vetula detrlt~m vidl, inque viginti fete g!ebulaa col. 'ld.hm, qu~ c~pfula artic~li comprehendebantur. Lig.~menmm ab zngulo maxiilz tnteriorls i~ n:,fculnm flytog!offam demi,~m ( r ) , htum~ membra~a~m, alterius nat~r~ eft.
VIIL
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Sump~bus S O C I E T A T I S M D C CL
N
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( m ) x ~ x l T I x t ! e ~ p, 8~, F. I~, L A~ (dem Wxn~r, ew pt. [ " ) p. 41o, A~ notehp¢ hg~men~mBt~.'r~r~ T, tL 1~ =;~, ~o) P~'r=e~tt, a~ua'~ attic P, 9,
m ¢~
TYPOG'RAPHIC~. X I V,
l-2a/g.r .Pf~fidq.¢.
Tom, V L
Fig. 1 Title page from Elementa Physiologiae Corporus Humani. Sumptibus Societatis Typographicae, Bernae 1764, Alberto von Hailer published by courtesy of the Librarian of The Royal Society of Medicine (Photographic Unit RSM).
Fig.2 von Halter's reference to loose bodies in the temporomandibular joint. (See text for translation)•
joint. On examination, the left joint was swollen, tender and crepitant. There was no limitation of mandibular movement. Radiographic examination confirmed osteoarthritic change, but not discernible loose bodies. Straw coloured synovial fluid was aspirated at operation and an arthrotomy disclosed eight small and irregular loose bodies. Recovery was uneventful and histological examination confirmed synovial chondromatosis.
joint, the left acromioclavicular joint and the left coracoid process (Fig. 8). An arthrotomy was performed and fiftytwo loose bodies removed (Fig. 9). The wound was drained and closed in layers. The patient was discharged on the 4th day operatively. The tissue was examined by three consultant pathologists and the following composite report has been given: Macroscopic appearance: The specimen consists of loose bodies from the left temporomandibular joint. There were 52 pieces of pale cartilaginous tissue weighing in aggregate 3.4 grams and varying in size from 3-15 mm. in maximum diameter and showing a smooth cobblestone surface. Microscopic appearance: The sections showed nodules of cartilaginous tissue, some with fibrinous surface tags, and two with quite extensive cystic degenerative change. Each had a squamoid epithelial surface. The cartilage cells were pleomorphic and many individual cells showed mineralization. No bizarre cells were present and mitoses were not seen. The central portions of the larger nodules had early mineralization of matrix as well as cartilage cells, but true bone formation was not present in these fragments.
Case 3. A 59-year-old woman was referred with a history of increased pain and swelling in the left temporomandibular joint resulting in mandibular deviation to the right and a left posterior open bite. The symptoms had developed over the preceding six months following a heavy fall in which she fractured her clavicle and damaged the left jaw joint. On examination there was an obvious preauricular swelling, 3 cm. in diameter, situated over the joint (Fig. 7). Tomographic examination confirmed both osteoarthritic changes and the presence of multiple radiopaque loose bodies. A bone study with single photon emission computed tomography (SPECT) was carried out with 800 MBq of technetium 99 m MDP. This bone scan was strongly positive for the affected temporomandibular
214
J. Cranio-Max.-Fac. Surg. 16 (1988)
Table 1
S u m m a r y of 42 c a s e s ( 1 7 6 4 - 1 9 8 7 ) w i t h t a b u l a t i o n of s a l i e n t p o i n t s
Paper
No.
Year
Sex Age Side Length of Synovial history in fluid years
Hailer 1 Axhausen 2
1764 1933
F M
21
R
Trevor Schneider ,~chneider Feist
3 4
1952 1960
F F
63 52
R R
4/12
5
1960
F
47
R
6
1960
F
59
Inovay
7
1962
F
Schulte
8
1969
F
J.E. de B. Norman et al.
Pain
Swelling
+
+
+
+
+
+
3
+
+
R
1
+
+
46
L
7/12
-
+
31
R
5
+
--
Crepitus
1 3
12 13
1972 1973
F F
37 53
L R
2/12 19
+
+ -
+
Tasanen 14
1974
F
40
R
5
+
-
+
2
Lomba
17
1977
F
32
R
1
Akhtar
18
1977
F
53
R
2
Ronald Ronald Ronald
19 20 21
1978 1978 1978
M M M
49 47 75
R
2 3/12 10/12
Thick clear amber Three aspirations
+
+
-
+
+
÷
÷
+
÷
+
+
A+C÷ Gelfoam
+ +
Ballard Ailing
L
Silastic A
1
4/12
62
A+C+
-
R
F
12x
4--.14
42
1977
1
7
M
16
+
+
1972
Raibley
+
Repeated dislocation, deviation Inability to close, deviation Deviation -
11
+
+
-
Ballard
cms) +
40~80
480
1
+
-
-
R
4/12
A+S
-
-
18
-
(2,5
A
-
-
M
L
10x 10x15 25x15
+
1971
44
1
+ Deviation +
-
+
1
+
+
(Concretion) 15
+
--
+
+
No histology No histology
2 Bodies le~ in situ Diagnosis benign synovialoma
A
3x2x 2~13 xl0x 3 25x10 x I0
A+C+S A
A
1)A 2)A+C Resection of tumour
29
4
Special features
A+C+M
8x5 0.5~4
10
F
+
-
Silver
1977
A+M+S
-
5
15
80
-
R
Rosen
-
+
-
28
+ (2.5 cms)
-
+
+
F
+
Multiple
+
1969
+ (2cms) -
A+S
+
-
9
+
-
-
+
Operations
20 Numerous 48
+
Kusen
Clear (8mls)
Limitation of Radiology Number Size manOsteo Loose loose mm. dibular arthritis bodies bodies movement
Diagnosis villonodular synovitis
A+S
5×5x 3410 x8 x5
1)SP 2)A
A+C A+C +
+
+
+
+
10
3~ lx17
+
20~30
245
A+C+M+ S+ Silastic A
+
25
1~5
A
+
1
+
1
Deviation
Ronald Ronald
Miller
22 23
1978 1978
F F
32 48
R
4/12 2
Cloudy (1 ml.)
+ +
+ +
+
Deviation
24
1978
F
61
L
10
Blenkin- 25 sopp
1978
M
60
L
1/12
Olley
26
1978
F
20
R
3/12
Fee
27
1979
F
48
R
1
÷
÷
Galliard 28 Takagi 29
1981 1981
F M
66 36
R R
6 3
--
+
+
+
+
+
Straw coloured Greenish gelati-
+ + (3 cms) ÷ + (3 cms)
--
--
+
nous
Deviation
-
-
+
(4x4 cms) Brants Morrish
36 31
1982 1983
M F
42 47
L L
25 6/12
+
Kraszewski
32
1984
F
49
R
6
÷
+
+
+
Deviation
--
÷
+ Deviation
+ (Scan +)
+
A
A+C+S+ Silastic 50 2~5 A 1)A+C Diagnosis(partial) villonodular 2 ) A + C synovitis (total) 70 A+S Several 3 ~ 1 5 A+C+ Gelfoam 107
20
A
Diagnosis osteochondritis dessicans
8ynovial Osteochondromatosis of the Temporomandibular Joint Table I
J. Cranio-Max.-Fac. Surg. 16 (1988)
215
Continued
Paper
No.
Year
Sex Age Side Length of Synovial Pain history in fluid years
Swelling
Crepitus
Limitation of Radiology Number Size manOsteo Loose loose ram. dibular arthritis bodies bodies movement
Operations
Kraszewski Blankestijn Btankestijn Blankestijn Oe Bont
33
1984
M
40
L
4
-
-
+
Deviation
A+M
34
1985
M
59
L
3/12
-
+
.
35
1985
M
19
R
18/12
+
+
-
36
1985
M
39
L
7
-
+
+
37
1985
F
63
R
4
Mucus
.
.
.
.
.
Deviation .
+
(Scan +) -
+ -
.
1+
40x30 x 30
20
1 ) A + SP
3
2) A
Numerous Multiple
A+C+S
Special features
A+C+ S
10
143.5
A+C+S
100
2x2x 542 x 2x8 1)SP 2)A+ S+M+ Silastic
A+M
U.C.H.
(Scan
+) Silver
38
1986
F
38
L
8/12
+
-
+
+
-
-
Thompson
39
1986
M
59
L
3
-
+ (2.5 x 3cms)
-
-
(Scan +)
Multiple2x3
Norman 40
1987
F
39
L
2
+
+
-
-
+
1
Norman
41
1987
F
39
L
1
+
+
-
+
-
8
9
Norman
42
1987
F
59
L
6/12
Straw + Coloured Straw + Coloured Straw + Coloured
+
52 (3.4g)
A+S
A C M S SP UCH
Arthrotomy Condylectomy Meniscectomy Synovectomy Superficial parotidectomy Unilateral condylar hyperplasia
= = = ~ ~ =
Scan + Blank
+ + (3cms)
= = = =
Left + Unilateral (Scan Open Bite, + ) Right Deviation
Technetium 99 m bone scan Present Not present Not mentioned
The periphery of the nodules was composed of fibrocartilage and one of the nodules had a peripheral focus of vascularity and cellularity which resembled granulation tissue (pannus formation). Diagnosis: temporomandibular joint - synovial (osteo) chondromatosis. Discussion Synovial choudromatosis may present with articular pain and preauricular swelling (mimicking a parotid tumour), crepitus and limitation of movement, with deviation to either side productive of a subtle degree of laterognathism. The swelling may be hard or fluctuant, and will probably move with the condyle or even disappear momentarily with mouth opening. A solitary loose body is rarely palpable, contrary to experience with larger joints. The mass does not transilluminate, and there is no associated muscle atrophy. Plain films and tomography of the joint (Figs. 3 and 4) may demonstrate the loose bodies, one or many (up to 480 have been described); and scintigraphy will usually show an area of increased uptake as illustrated in the accompanying photographs (Figs. 5 and 6). The use of CT scanning and in cases of diagnostic dubiety CT sialography is not disputed, and will clearly be of immeasurable value in cases where the mass is thought to arise from the parotid gland. Preliminary diagnostic arthroscopy would seem to be unnecessary in the presence of a good Mstory, physical and radiographic findings.
DiagnosisMixed Parotid Tumour C+S
U.C.H.
A+8
summary female patients male patients right side left side side not stated
28 14 22 16 4
Joint Fluid There may be an obvious effusion, although the state of the synovial fluid has not been the object of comment in most of the reported cases. In our three examples the synovial fluid was straw coloured, and in each instance it was of increased volume and viscosity, albeit not fibrin flecked. In the third case, there were a few pus cells present and the Rheumaton test of the fluid was negative.
Nodules The nodules may be pearly white, grey, or of a pink or light azure hue. They are of variegated shape and ranging in size from 1 mm. to 1.5 cm. or larger, and in the latter case may conform to the configuration of the caput, the fossa, or both. The meniscus or disc may be absent in such instances and when present in our cases it was not perforated. The synovium is generally thickened and the glenoid fossa and articular eminence denuded or even studded with diminutive nodules in various stages of development, including some of petiolar form. This was observed in the most recent arthrotomy of the series and the embryonic nodules were, for the most part, readily detached from the vault with a Volkmann's spoon. Conglomerate mass lesions of osteocartilaginous material were not encountered in this series, although they have been described. Multiple synovial chondromata may be found in tendon sheaths, bursae and at a distance from the joint (Geshickter and Copeland, 1949; Lichtenstein, 1972). Only one para-articular chondroma arising from the tern-
216
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Fig. 3 Sagittal tomogram of the left temporomandibular joint showing enlargement of the joint space, forward positioning of the caput in addition to an aggregate of loose bodies,
Fig.5 Single photon emission computed tomography (SPECT) of skull and jaw with 800 MBq Tc99 m MDP which is strongly positive for the left temporomandibularjoint (image 12).
J.E. de B. Norman et al.
Fig.4 Coronal tomogram of the same joint showing lamellar structure of loose body.
Fig.6 Radionuclide scan (SPECT) showing identical findings (image22).
poromandibular joint, and invading the parotid gland, has been seen in over three hundred operations on the jaw joint and a further combined series of two hundred and fifty turnouts of the major and minor salivary glands (Norman and Williams, 1984). The same series includes a solitary synovial cyst arising from the temporomandibular joint and invading the parotid, presenting as a salivary gland mass and requiring superficial parotidectomy and facial nerve dissection. Diagnostic confusion may occur, and it is educative to review the thoughtful paper by Thompson et al. (1986) in which an intra parotid mass was first thought to be a pleomorphic salivary adenoma and later an example of synov-
Fig.7 Photograph of an obvious preauricular swelling.
ial chondromatosis. We suggest that the initial lesion had the characteristic appearance of a para-articular chondroma and was remarkably similar to a case in our own series.
Pathogenesis of Loose Bodies Synovial chondromatosis has been described in primary and secondary forms. Primary the synovium develops from the embryonic mesenchyme and as a consequence of metaplasia, cartilaginous foci develop within the synovial membrane. An intra-synovial spherical body slowly forms with the villous process of the synovial membrane as its pedicle. This later becomes
Synovial Osteochondromatosisof the TemporomandibularJoint
J. Cranio-Max.-Fac.Surg. 16 (1988)
217
Fig.8 Radionuclide scan (SPECT) of same patient strongly positive for left acromioclavicular joint and left coracoid process (recent trauma).
Fig. 9 Intraoperative photograph showing extent of the arthrotomy and accumulation of multiple loose bodies displayed on an huckaback towel,
detached and falls into the joint cavity where it becomes enveloped by a perichondrium and is nourished by diffusion of the synovial fluid. The chondroblasts and osteoblasts proliferate and enlargement occurs by surface layering. Resorption can occur as the result of osteoclastic activity and may produce surface irregularity. Necrosis of the central area may occur as the result of deprivation of synovial nutrition. Although calcification may follow in this zone, re-attachment and re-vascularization have been described. Dahlin (19 81) reports that "histologic appearance of these proliferating tumours is what makes them important to the pathologist. In many instances the nuclear abnormalities and numbers of multinucleated cells are so great that in the right context, as in the medulla of a major tubular bone, the diagnosis of chondrosarcoma would be strongly supported. Paradoxically, however, these cartilaginous masses are practically never malignant nor even premalignant." This is an active metaplastic rather than neoplastic process without apparent underlying disease. Milgram (1977 a, b, c) recognizes three phases: (a) Active synovial disease without loose bodies. (b) Synovial nodules with loose bodies. (c) Multiple loose bodies with inactive synovial disease. The number of loose bodies varies and the record of 1047 in a single joint appeared to be held by Berry (1891, 1894) until we came upon Bland-Sutton's report (1907) of 1532 loose bodies in a shoulder joint. Our modest 52 loose bodies pales into insignificance. Bilaterality is rare, albeit described by Humpbry (1888) in relation to the elbow joint. The first unilateral case described in the Australian literature may have been by Syme (1888). It rarely occurs in children and the youngest patients in
those cases involving large joints were 5 and 8 years respectively. Secondary synoviaI chondromatosis is a more passive process involving synovial nourishment of dislodged fragments of joint tissue. The latter may be secondary to trauma or to joint disease such as osteochondritis dessicans and inflammatory or non inflammatory arthropathy. The dislodged bone or cartilage fragments undergo concentric layering. They may lodge in the synovium with consequent cartilaginous metaplasia and nodule formation which is more irregular than that of primary chondromatosis and shows no cytological atypia.
Treatment "The treatment is essentially surgical. In the literature we have noted such radical procedures as amputation, resection and synovectomy. We have removed the bodies by the simplest possible route, and as recurrences have been so rare we see no need to change our procedure." Henderson and Jones (1923) The observation made by two orthopaedic surgeons from the Mayo Clinic 64 years ago in relation to procedures upon large weight bearing joints remains apposite (Jones, 1924). The management of the condition is surgical and whilst we have no doubt that removal of loose bodies from the jaw joint via the operating arthroscope may be the technique of the future, the delicate nature of the needle arthroscopes presently in use precludes all but the smallest joint mice being retrieved per arthroscopy. Arthrotomy, therefore, is indicated and the joint exposed via a standard temporofacial incision as described elsewhere (AI-Kayat and Bramley, 1979; Norman, 1982). Comfortable surgical access and working space is essential as the furthest reaches of the joint need to be examined
218
J. Cranio-Max.-Fac. Surg. 16 (1988)
Fig. 10 Photomicrograph of loose body (case 3) with dense cartilage and an early mineralized margin contained within synovium. (HE x20).
Fig, 12 Photomicrograph of cartilage island surrounded by fibrin and embedded in synovium. Some of the cartilage cells are pleomorphic and the stroma is very slightly mineralized (per kind favour of Mr. A. Shehata and Dr. R. Holt). (HE ×40). '
and any occult loose body removed. The authors favour the temporofacial incision, with splitting of the temporalis muscle to allow relaxed forward dissection along the zygomatic arch at the subperiosteal level. The glenoid lobe of the parotid gland and the contained temporofacial ramus of the seventh cranial nerve are swept forward from the joint capsule with a Dickson-Wright dissector. Where an effusion is encountered or loose bodies are anticipated, an aspiration is carried out prior to arthrotomy and the synovial fluid submitted for examination. Upon incising the joint capsule, the surgeon will almost certainly be rewarded by a flow of viscid synovial fluid followed by the egress of many pearly white bodies, some smooth, some slightly faceted and some bossellated. A thorough examination of the joint will require forward displacement of the condyle (by the assistant) in order that every nook and cranny be inspected. Movement of the condyle and gentle massaging of the overlying tissues will produce further bodies and this condylar movement and massage is considered desirable in order that any material
J.E. de B. Norman et al.
F i g . l l Multiple cartilage islands, some with pleomorphic cells, embedded in synovium (per kind favour of Mr. A. Shehata and Dr. R. Holt). (HE x 20).
still adherent by a footstalk will be detached. The joint is repeatedly irrigated and inspected. The surgeon should not refrain from a limited synovectomy and must carefully inspect the vault. In the third patient we noted numerous embryonic bodies in this area, a few of which were attached by a pedicle, and this zone was scarified with a Volkrnann's spoon. It is unnecessary to remove the mandibular condyle for this benign condition, although a limited synovectomy is advisable. We consider that a total synovectomy is impossible of achievement with the mandibular condyle in situ, and there is no valid indication to remove it. The authors recommend a partial or limited synovectomy and the accessible synovial lining is dissected out with fine micro scissors (Owen-Vickers type) and bleeding points are sealed with the bipolar diathermy. The procedure is carried out under magnification. At the conclusion of the operation, the surgeon may wish to inspect the furthest reaches of the joint with the arthroscope and we have employed the Wolf Lumina SL 1.9 ram. telescope over the triennium for diagnostic arthroscopy. Following meticulous haemostasis, the wound is carefully closed in layers with suction drainage. A temporo-mastoid dressing is applied and the patient discharged on the 4th post-operative day to be followed up at the out-patient clinic. A puree diet is advised for the first week and normal sustenance thereafter.
Conclusions An historical review of synovial osteochondromatosis affecting the temporomandibular joints has been presented with a review of the relevant literature. Although the condition is of singular rarity, the physical signs are relatively characteristic and include pain, swelling and crepitus. Plain films may not adequately demonstrate the loose bodies and the clinician may need to resort to sagittal tomography or even CT scanning of the joint. The treatment of synovial chondromatosis is surgical and the loose bodies should be removed by arthrotomy with meticulous examination of the joint cavity. We consider it unnecessary to
Synovial Osteochondromatosis o f the Temporomandibular Joint remove the m a n d i b u l a r condyle for this benign condition, although a limited synovectomy m a y be advisable. Acknowledgements Appreciation and thanks are due to Professor B.E.D.Cooke, FRCPath., for advice in the preparation of this paper. The photomicrographs illustrating an embryonic form of osteochondromatosis from the finger joint are published by kind permission of Dr, R.S. Hoh, DCP (Lond.), and Mr. N.G.Shehata, FRCS., FRACS, Thanks are due to Mrs. Elizabeth de Kantzow, BA (Syd.), Grad. Dip. Lib. Sc., ALAA., for constructive criticism in the preparation of the text and bibliography. The scholarship of Ronald A. Elliott, Esq., BA (Syd.), in translating the Latin text is acknowledged with pleasure. The photographs have been taken by Mr. Timothy CoIlis-Bird, and the manuscript typed by Mrs. Jocelyn Hungerford and Miss Gabriella Kalmar.
References
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Syme, G. A.: A case of loose body removed from the elbow-joint, presumably due to fracture of the head of the radius. Aust. Med. J. 10 (1888) 323 Takagi, M., G. Ishihawa: Simultaneous villonodular synovitis and synovial chondromatosis of the temporomandibular joint: Report of case. J. Oral Surg. 39 (1981) 699 Tasanen, A., M. A. Lamberg, R. Kotilainen: Osteochondromatosis of the temporomandibular joint. Oral Surg. 38 (1974) 845 Thompson, K., H. C. Schwartz, J. W. Miles: Synovial chondromatosis of the temporomandibular joint presenting as a parotid mass:
J. E. de B. N o r m a n et al.: Synovial Osteochondromatosis possibility of confusion with benign mixed tumor. Oral Surg. 62 (1986) 377 Trevor, D.: A case of synovial chondromatosis of the temporomandibular joint. Postgrad Med. J. 28 (1952) 408
Mr. J. E. de B. Norman, FRCS (Ed.) 9 West Street, Hurstville New South Wales, 2220 Australia