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Synovial chondromatosis of temporomandibular joint
Section
of the
Federal dental services
Synovial chondromatosis of temporomandibular joint Report
of a case
William C. Schulte, Colonel, USAF (DC),* R.obert R. Rhyne, Major, UXAF (DC)**
am8
USAF
BASE,
HOSPI’TAL,
WRIGHT-PATTERSON
AIR
FORCE
DAYTON,
OHIO
S
ynovial chrondromatosis is a benign pathosis characterized by the development of metaplastic cartilaginous foci within the synovial membranes of articulating joints. Lannec first described this condition in 1813,l and Brodie classified it as neoplasia in 1846.l Although the cause of the disease is unknown, most experts in osseous pathology believe that the process is not one of a “blastic” type of growth but, rather, cartilaginous metaplasia of primitive skeletogenic mesenchymal tissue remnants in the synovial membrane. The metaplastic transformation to cartilage has been likened to a similar condition, myositis ossificans, in which cartilaginous and osseous tissues develop in skeletal muscle following trauma ; however, the role of trauma as the precipitating factor in chondromatosis is still debated.2 Jaffr? has stated that, once the metaplastic foci of cartilage has developed, the nodules grow by active multiplication of the cartilage cells within the metaplastic foci, rather than through simple accretion of cartilage by additional metaplasia of mesenchymal remnant.s. As the nodules increase in size, they may, The contents of this article are the personal views of the authors and are not to be construed as a statement of official Air Force policy or as an Air Force endorsement of any commercial product described. *Chief of Oral Surgery and Chief of Professional Services (Dental Division). **Formerly Clinical Instructor in Oral Pathology, University of Missouri at Kansas City, School of Dentistry.
906
Volume 28 Number 6
Synovial
chon.dromat0si.s of temporomandibulw
joint
907
by physical force of the multiplying cartilage cells, push into the surrounding synovial connective tissue, giving the impression of fibrous encapsulation. As further multiplication occurs, the nodules may protrude into the affected joint space and become detached, presumably by trauma. The loose body or bodies, sometimes referred to as “joint mice,” may continue to grow and increase in size, receiving their nourishment from the surrounding synovial fluid of the joint. Large bodies may seriously affect joint movement, and this usually is the first subjective sign of the disorder. Synovial chondromatosis is rare,” and it is indeed a rare occurrence in the t,emporomandibular joint. Although many suspected cases of synovial chondromatosis have been reported, Jaffe* has encountered only six cases of true synovial chondromatosis, with the knee being the predilected site. Aegerter and KirkPatrick3 have expressed the opinion that, since the process is entirely nonneoplastic, synoviul chcrulromatosis is a poor descriptive term and should be reserved for true osteochondroma; they suggest the term periarticular tenosynovial chondrometaplasia. Other articulating joint diseases with loose bodies in the affected space can, both clinically and radiographically, simulate synovial chondromatosis. Such diseases include degenerative joint disease, rheumatoid arthritis, neurotrophic arthritis, tubereulous arthritis, osteochondritis dissecans, and osteochondral fractures. However, if the exacting criteria of metaplastic cartilaginous development and multiplication from the synovial membrane are used, then the other conditions cannot be diagnosed correctly as synovial chondromatosis. An extensive study4 conducted at the Mayo Clinic of thirty-two cases diagnosed as synovial chondromatosis during the period from 1910 to 1957 revealed an age range of 16 to 67 years, with an average of 40 years. Twenty-one of the thirty-two cases occurred in men, and all were mono-articular. The three main predileeted sites were the knee (22)) the hip (5)) and the elbow (2). No mention was made of incidence in the temporomandibular joint. Subjective symptoms were notably pain, swelling, tenderness, and limited motion of the affected joint. The duration of symptoms ranged from 1 month to 47 years, with an average of 5.5 years. In thirteen of the thirty-two cases there was evidence of radiopaque masses in the joint space ranging from 5 to 15 mm. in size. Another paramount point in the diagnosis of synovial chondromatosis is the notable cellularity of the cartilage foci in histologic sections. As previously mentioned, growth is from multiplication of the metaplastic cells and not from accretion by a,dditional metaplasia ; thus, the tissue may be incorrectly diagnosed as chondrosarcoma if the only factor considered is the cell morphology. The presence of large multinucleated plump pleomorphic cells is in reality an index of actively proliferating benign cartilage cells. Jaffe’ has stated that “a lesion not contained within ;I bone, even though histologically it is cellular, is not indicative that the lesion is no longer benign.” This point should be emphasized because of the treatment procedures of the two diseases. Treatment of synovial chondromatosis involves surgical removal of the loose body and synovectomy as much as possible. Treatment of chondrosarcoma involves a radical surgical procedure and possibly resultant severe disfigurement.
908
Schulte
and Rhyne
OS., O.M. & 0.1’. December, 1969
CASE REPORT A 31-year-old Caucasian woman was admitted to the hospital for the second time on Aug. 9, 1966, with a history of arthritis of the right tenlporom:~ndil)ular joint and limitation of functional movement. The condition had been present for about 4 to 5 years, with increasing limitation of function and pain in the right temporomandibular joint.. Onset was noticed to be associated with a tremendous yawn and “popping” of the right joint,. Subsequent to that episode, symptoms of stiffness and pain in the right temporomandibular joint gradually manifested themselves, becoming more acutely excruciating in the past year. Previous radiographic studies at the time of the first admission in February, 19ti5, revealed degenerative changes in the right temporomandibular joint and the presence of a loose body in the posterior region of the glenoid fossa. At that time, the patient had a 2 cm. maximum range of jam opening, measured at the incisal edges of the maxillary and mandibular incisors. This interincisal distance had diminished to 1 cm. at the time of the second admission some 18 months later. Past history
The patient was a dependent housewife, who had been married for 10 years. She denied the use of tobacco but said that she drank an occasional social cocktail. She XV= now taking the usual prenatal vitamins, including iron, in association with her current pregnancy. The patient’s 52.year-old father had a history of arthritis for the past 5 or 6 years. The family history was otherwise not contributory. The patient sustained the usual childhood diseases without sequelae. She had had no serious illnesses during her adult life. She had suffered a dislocation of the left hip at the age of 5 years; this was treated without any residual problem or limitation of function. The patient had no known allergies or drug sensitivities. Review
of systems
Findings were essentially not remarkable except that during the past 2 years the patient had suffered frequent headaches in the right temporal and frontal regions in association with The patient was pregnant at approximately the right temporomandibular joint arthritis. 6 months in term. The review of systems was otherwise not remarkable. Physical
examination
Upon admission to the hospital, the patient’s blood pressure was 120/84, with a pulse rate of 86. Examination revealed a well-nourished, well-developed pregnant woman in no acute distress but with a chronic problem of pain and limitation of motion in the right temporomandibular joint. Examination of the head and neck revealed that the patient was normocephalic. The thyroid was not palpable. The pupils reacted equally to light accommodation. The fundi were normal. The tympanic membranes were clear. The patient could not move the jaw in protrusion or open the mouth beyond 1 cm. Opening efforts precipitated marked pain in the right temporomandibular joint. There was no tenderness to palpation of the chest revealed good symover the temporomandibular joint, however. Examination metrical bilateral expansion; the chest was clear to percussion and auscultation. The sinus rate and rhythm were regular. The patient had a Grade 1 systolic ejection murmur at the left base. The point of maxilla impulse was at the fifth intercostal space. The breasts were normal ; no masses were palpable. Examination of the abdomen revealed no masses or organomegaly. There was no evidence of hernia. The uterus appeared to be 6 months in size and nontender. The extremities mere normal, with no pitting edema. Deep tendon reflexes were physiologic. Physical examination findings were otherwise not remarkable. Our impression was that this was a case of degenerative disease of the right temporomandibular joint with partial ankylosis. Laboratory
data
On admission to the hospital, the patient’s white blood cell count was 10,800, with 74 per cent neutrophils, 3 per cent band cells, 17 per cent lymphocytes, 3 per cent monooytes,
Synovial chondromatosis
of temporomandibdar
joint
909
and 3 per cent eosinophils. The hematocrit was 39 per cent, and the hemoglobin was 13.0 Gm. Bleeding and coagulation times were within normal limits. Blood morphology was normal. Urinalysis revealed a ~1% of 6 and a specific gravity of 1.005; albumin, sugar, and acetone were negative Radiographic
tlndings
A radiograph of the chest showed no significant abnormalities of the heart and lungs. Radiographic tomography of the temporomandibular joints was performed in both the open and closed positions in the lateral projection. Advanced degenerative changes mere noted in the right temporomandibular joint; these included pseudocystic lesions on the articular surface of the condyloid process and posterior slope to the articular eminence. There was also evidence of sclerotic changes on the posterior surface and crest of the articular eminence
E
E
Pig. 1. Presurgical tomogram of right temporomandibular joint illustrating loose body within joint space (arrows). Fig. &. Presurgical tomogram of left temporomandibular joint illustrating normal-appearing joint.
910
Schdte
and Rhync
as well as the condylar head. Marked narrowing ef the joint space was also evident. There was a large radiopaque loose body situated in the posterior half of the right temporomandibular joint, measuring approximately 5 11y 11 mm. (Fig. 1). The open-mouth view revealed marked limitation in excursion of the condyloitl process. Examination of the left temporomandibular joint revealed a normal articulation (Fig. 2j. However, in the openmouth views evidence of limitation of excursion of the left condgloid proccss was noted and was thought to be secondary to the functional limitation imposed by the partially ankylosed right temporomandibular joint. Comparison with the previous (1965) radiographic cxamination revealed marked progression of degencrativc disease in the right temporomandibular joint. of degenerative disease of The radiologist’s diagnostic impression was “marked progression tho right tcmporomandibular joint, with scvcre limitation of motion and prcsencc of a loox body within the right joint.”
3
Fig. 4
Pig. 3. Nidus of proliferating pleomorphic hyperchromatic cartilage cells from lo connecl live bod .Y in right temporomandibular joint space n-ith surrounding dense hyalinized tiss UP. Fig. 4. Medium-power view of specimen demonstrating remnants of synovium, , hyalini zed con nective tissue, and metaplastic cartilage.
SynoGdd Surgical
chomiromatosis
of tentporo~nd2ruZa.r
jokt
911
procedure
With the patient under gtmeral anesthesia, an arthrotomy of the right temporomandibular joint, including removal of t:re loose body and a high condylectomy, was performed from a preauricular incision. A Silastic implant was positioned in the joint space in the region occupied by tho resected condylar head. This implant was placed to prevent the possible development of ankylosis of the joint. The wound was then closed in layers in the usual manner. The patient tolerated the procedure extremely well, and postoperative recovery was uneventful. On the first postoperativl : day the patient was taking a full liquid diet without difficulty and had only moderate pain or discomfort. There was minimal edema associated with the site of the preauricular incision. On the third postoperative day sutures were removed from the preauricular incision, and the wound was observed to be healing well. At this time the patient was placed on a dental soft diet and started on a regimen of exercise to increase the maximum range of open’ng of the jaws. The patient continued to progress well without any marked degree of pain cmrdiscomfort, and by the fifth postoperative day she was able to open the jaws 2.5 cm., measured at the incisal edges of the anterior teeth. The patient was continued on an exercise routine and within 3 weeks was handling a regular diet well. She was discharged from the hospital on Aug. 19, 1966, to return home. Histopathology
Three specimens (HPS-490566 ABC) were submitted for histologic study and diagnosis. Specimen A, the loose body in the joint, measured 12 by 8 1)~ 5 mm., and gross examination revealed the specimen to be consistent with cartilage tissue. Specimen B, labeled “meniscus,” consisted of multiple fragments of tissue compatible with cartilage and fibrous tissue. Specimen B had a total volume of 3 ml. Specimen C was an irregular fragment of bone (condylar head) with a flat surface denoting that it had been surgically removed. This specimen measured 12 by G by 6 m:n. Sections of specimens A and B (loose body and meniscusj consisted primarily of sheets and clumps of actively grooving cartilage which were surrounded by dense hyalinized connective tissues (Fig. 3). Scattered remnants of synovial tissue were evident within the thickened connective tissue (Fig. 4). In many areas, there were transitional zones in which the connective tissue cells were plump and undergoing cartilaginous metaplasia (Fig. 5). The lesion was somewhat disturbing morphologically, in that the majority of cartilage cells were binucleate and multinucleate with hyperchromatic nuclei (Figs. 3 and 6). This pleomorphism is to be interpreted as actively growing cartilage and not as an index of malignant growth. Discrete areas of calcification and ossification were evident throughout the specimen (Fig. 7). Sections of specimen C consisted of relatively sclerotic bone tissue with a normal-appearing mature cartilaginous cap.
DISCUSSION
Although reports of chondrosarcoms arising from areas of synovial chondromatosis have been reported, we have been able to locate only one reported case of chondrosarcoma which developed from a previously radiographically and histologically diagnosed case of synovial chondromatosis.” It is postulated that in the other reported (:ases the lesions were actually chondrosarcoma from the start. Because of the morphology of the cells in chondromatosis, this condition could understandably be misdiagnosed as chondrosarcoma if one used only the histologic criteria. Therefore, a critical evaluation of the clinical and mdiographic evidence must be performed in arriving at a definite diagnosis. In this particular case, the lesions were evident for well over 18 months with no additional large appreciable areas of “Iytic” or “bIastic” degeneration in the area of the temporomandibular joint which, if present., would have been interpreted as a malignant, rapidly growing tumor. The preoperative diagnosis was one of benign degenerative arthritis. Only upon histologic examination of the tissue
912
Xchulte
08, O.M. & O.P. leic mher, 1 968
and Rhwns
Fig. 5
Fig.
Fig. cartilage. Fig. Fig. areas of
6
5. High-power
view
showing
metaplastio
transformation
of synovisl
remnants
into
6. High-power 7. High-power calcification.
view of typical cartilage cells of surgically removed specimens. view of loose body demonstrating pleomorphic cartilage cells and
Volume 28 Number 6
SynoGal
chondromatosis
of temporomandibular
joint
913
did the suspicion of chondrosarcoma arise, and only after extensive evaluation of the histologic features, behavior pattern of the tumor, subjective symptoms, and radiographs was the diagnosis of synovial chondromatosis established. The postoperative follow-up has been difficult, as the patient has moved to another geographic area. A personal letter confirms that she is free of symptoms at the present time and has as :Cunctional a temporomandibular joint as at any time in her life. SUMMARY
A single case of synovial chondromatosis has been presented. The disease entity is rare, when all articulating joints are considered, but the involvement of the temporomandibular joint is practically nonexistent. Although synovial chondromatosis is a benign process, the bizarre morphologic appearance of the tissue cells presents a difficult diagnostic problem in distinguishing between chondromatosis and chondrosarcoma. Management of the patient is, indeed, less dramatic and traumatic in the case of chondromatosis than when an erroneous diagnosis of chondrosarcoma is made. It is essential that radiologic, histologic, and clinical expressions be considered before a final diagnosis is made and surgical treatment is instigated. REFERENCES
1. Luck, V. C.: Bone and Joint Diseases, Springfield, Ill., 1950, Charles C Thomas, Publisher, pp. 576-579. 2. Jaffe, H. L. : Tumors and Tumorous Conditions of the Bones and Joints, Philadelphia, 1961, Lea & Febiger, pp. Em58-567. 3. Aegerter, E., and Eirkpa.trick J.: Orthopedic Diseases, ed. 2, Philadelphia, 1963, W. B. Saunders Company, p. 767. 4. Murphy, F. P., Dahlin, .D. C., and Sullivan, C. R.: Articular Synovial Chondromatosis, J. Bone & Joint Surg. 444.: 77-86, 19632. 5. Mullins, F., Berard, C. -W., and Eisenberg, 5. H.: Chondrosarcoma Following Synovial Chondromatosis, Cancer X3: 1180-1188, 1965.
of the
Federal dental services
Synovial chondromatosis of temporomandibular joint Report
of a case
William C. Schulte, Colonel, USAF (DC),* R.obert R. Rhyne, Major, UXAF (DC)**
am8
USAF
BASE,
HOSPI’TAL,
WRIGHT-PATTERSON
AIR
FORCE
DAYTON,
OHIO
S
ynovial chrondromatosis is a benign pathosis characterized by the development of metaplastic cartilaginous foci within the synovial membranes of articulating joints. Lannec first described this condition in 1813,l and Brodie classified it as neoplasia in 1846.l Although the cause of the disease is unknown, most experts in osseous pathology believe that the process is not one of a “blastic” type of growth but, rather, cartilaginous metaplasia of primitive skeletogenic mesenchymal tissue remnants in the synovial membrane. The metaplastic transformation to cartilage has been likened to a similar condition, myositis ossificans, in which cartilaginous and osseous tissues develop in skeletal muscle following trauma ; however, the role of trauma as the precipitating factor in chondromatosis is still debated.2 Jaffr? has stated that, once the metaplastic foci of cartilage has developed, the nodules grow by active multiplication of the cartilage cells within the metaplastic foci, rather than through simple accretion of cartilage by additional metaplasia of mesenchymal remnant.s. As the nodules increase in size, they may, The contents of this article are the personal views of the authors and are not to be construed as a statement of official Air Force policy or as an Air Force endorsement of any commercial product described. *Chief of Oral Surgery and Chief of Professional Services (Dental Division). **Formerly Clinical Instructor in Oral Pathology, University of Missouri at Kansas City, School of Dentistry.
906
Volume 28 Number 6
Synovial
chon.dromat0si.s of temporomandibulw
joint
907
by physical force of the multiplying cartilage cells, push into the surrounding synovial connective tissue, giving the impression of fibrous encapsulation. As further multiplication occurs, the nodules may protrude into the affected joint space and become detached, presumably by trauma. The loose body or bodies, sometimes referred to as “joint mice,” may continue to grow and increase in size, receiving their nourishment from the surrounding synovial fluid of the joint. Large bodies may seriously affect joint movement, and this usually is the first subjective sign of the disorder. Synovial chondromatosis is rare,” and it is indeed a rare occurrence in the t,emporomandibular joint. Although many suspected cases of synovial chondromatosis have been reported, Jaffe* has encountered only six cases of true synovial chondromatosis, with the knee being the predilected site. Aegerter and KirkPatrick3 have expressed the opinion that, since the process is entirely nonneoplastic, synoviul chcrulromatosis is a poor descriptive term and should be reserved for true osteochondroma; they suggest the term periarticular tenosynovial chondrometaplasia. Other articulating joint diseases with loose bodies in the affected space can, both clinically and radiographically, simulate synovial chondromatosis. Such diseases include degenerative joint disease, rheumatoid arthritis, neurotrophic arthritis, tubereulous arthritis, osteochondritis dissecans, and osteochondral fractures. However, if the exacting criteria of metaplastic cartilaginous development and multiplication from the synovial membrane are used, then the other conditions cannot be diagnosed correctly as synovial chondromatosis. An extensive study4 conducted at the Mayo Clinic of thirty-two cases diagnosed as synovial chondromatosis during the period from 1910 to 1957 revealed an age range of 16 to 67 years, with an average of 40 years. Twenty-one of the thirty-two cases occurred in men, and all were mono-articular. The three main predileeted sites were the knee (22)) the hip (5)) and the elbow (2). No mention was made of incidence in the temporomandibular joint. Subjective symptoms were notably pain, swelling, tenderness, and limited motion of the affected joint. The duration of symptoms ranged from 1 month to 47 years, with an average of 5.5 years. In thirteen of the thirty-two cases there was evidence of radiopaque masses in the joint space ranging from 5 to 15 mm. in size. Another paramount point in the diagnosis of synovial chondromatosis is the notable cellularity of the cartilage foci in histologic sections. As previously mentioned, growth is from multiplication of the metaplastic cells and not from accretion by a,dditional metaplasia ; thus, the tissue may be incorrectly diagnosed as chondrosarcoma if the only factor considered is the cell morphology. The presence of large multinucleated plump pleomorphic cells is in reality an index of actively proliferating benign cartilage cells. Jaffe’ has stated that “a lesion not contained within ;I bone, even though histologically it is cellular, is not indicative that the lesion is no longer benign.” This point should be emphasized because of the treatment procedures of the two diseases. Treatment of synovial chondromatosis involves surgical removal of the loose body and synovectomy as much as possible. Treatment of chondrosarcoma involves a radical surgical procedure and possibly resultant severe disfigurement.
908
Schulte
and Rhyne
OS., O.M. & 0.1’. December, 1969
CASE REPORT A 31-year-old Caucasian woman was admitted to the hospital for the second time on Aug. 9, 1966, with a history of arthritis of the right tenlporom:~ndil)ular joint and limitation of functional movement. The condition had been present for about 4 to 5 years, with increasing limitation of function and pain in the right temporomandibular joint.. Onset was noticed to be associated with a tremendous yawn and “popping” of the right joint,. Subsequent to that episode, symptoms of stiffness and pain in the right temporomandibular joint gradually manifested themselves, becoming more acutely excruciating in the past year. Previous radiographic studies at the time of the first admission in February, 19ti5, revealed degenerative changes in the right temporomandibular joint and the presence of a loose body in the posterior region of the glenoid fossa. At that time, the patient had a 2 cm. maximum range of jam opening, measured at the incisal edges of the maxillary and mandibular incisors. This interincisal distance had diminished to 1 cm. at the time of the second admission some 18 months later. Past history
The patient was a dependent housewife, who had been married for 10 years. She denied the use of tobacco but said that she drank an occasional social cocktail. She XV= now taking the usual prenatal vitamins, including iron, in association with her current pregnancy. The patient’s 52.year-old father had a history of arthritis for the past 5 or 6 years. The family history was otherwise not contributory. The patient sustained the usual childhood diseases without sequelae. She had had no serious illnesses during her adult life. She had suffered a dislocation of the left hip at the age of 5 years; this was treated without any residual problem or limitation of function. The patient had no known allergies or drug sensitivities. Review
of systems
Findings were essentially not remarkable except that during the past 2 years the patient had suffered frequent headaches in the right temporal and frontal regions in association with The patient was pregnant at approximately the right temporomandibular joint arthritis. 6 months in term. The review of systems was otherwise not remarkable. Physical
examination
Upon admission to the hospital, the patient’s blood pressure was 120/84, with a pulse rate of 86. Examination revealed a well-nourished, well-developed pregnant woman in no acute distress but with a chronic problem of pain and limitation of motion in the right temporomandibular joint. Examination of the head and neck revealed that the patient was normocephalic. The thyroid was not palpable. The pupils reacted equally to light accommodation. The fundi were normal. The tympanic membranes were clear. The patient could not move the jaw in protrusion or open the mouth beyond 1 cm. Opening efforts precipitated marked pain in the right temporomandibular joint. There was no tenderness to palpation of the chest revealed good symover the temporomandibular joint, however. Examination metrical bilateral expansion; the chest was clear to percussion and auscultation. The sinus rate and rhythm were regular. The patient had a Grade 1 systolic ejection murmur at the left base. The point of maxilla impulse was at the fifth intercostal space. The breasts were normal ; no masses were palpable. Examination of the abdomen revealed no masses or organomegaly. There was no evidence of hernia. The uterus appeared to be 6 months in size and nontender. The extremities mere normal, with no pitting edema. Deep tendon reflexes were physiologic. Physical examination findings were otherwise not remarkable. Our impression was that this was a case of degenerative disease of the right temporomandibular joint with partial ankylosis. Laboratory
data
On admission to the hospital, the patient’s white blood cell count was 10,800, with 74 per cent neutrophils, 3 per cent band cells, 17 per cent lymphocytes, 3 per cent monooytes,
Synovial chondromatosis
of temporomandibdar
joint
909
and 3 per cent eosinophils. The hematocrit was 39 per cent, and the hemoglobin was 13.0 Gm. Bleeding and coagulation times were within normal limits. Blood morphology was normal. Urinalysis revealed a ~1% of 6 and a specific gravity of 1.005; albumin, sugar, and acetone were negative Radiographic
tlndings
A radiograph of the chest showed no significant abnormalities of the heart and lungs. Radiographic tomography of the temporomandibular joints was performed in both the open and closed positions in the lateral projection. Advanced degenerative changes mere noted in the right temporomandibular joint; these included pseudocystic lesions on the articular surface of the condyloid process and posterior slope to the articular eminence. There was also evidence of sclerotic changes on the posterior surface and crest of the articular eminence
E
E
Pig. 1. Presurgical tomogram of right temporomandibular joint illustrating loose body within joint space (arrows). Fig. &. Presurgical tomogram of left temporomandibular joint illustrating normal-appearing joint.
910
Schdte
and Rhync
as well as the condylar head. Marked narrowing ef the joint space was also evident. There was a large radiopaque loose body situated in the posterior half of the right temporomandibular joint, measuring approximately 5 11y 11 mm. (Fig. 1). The open-mouth view revealed marked limitation in excursion of the condyloitl process. Examination of the left temporomandibular joint revealed a normal articulation (Fig. 2j. However, in the openmouth views evidence of limitation of excursion of the left condgloid proccss was noted and was thought to be secondary to the functional limitation imposed by the partially ankylosed right temporomandibular joint. Comparison with the previous (1965) radiographic cxamination revealed marked progression of degencrativc disease in the right temporomandibular joint. of degenerative disease of The radiologist’s diagnostic impression was “marked progression tho right tcmporomandibular joint, with scvcre limitation of motion and prcsencc of a loox body within the right joint.”
3
Fig. 4
Pig. 3. Nidus of proliferating pleomorphic hyperchromatic cartilage cells from lo connecl live bod .Y in right temporomandibular joint space n-ith surrounding dense hyalinized tiss UP. Fig. 4. Medium-power view of specimen demonstrating remnants of synovium, , hyalini zed con nective tissue, and metaplastic cartilage.
SynoGdd Surgical
chomiromatosis
of tentporo~nd2ruZa.r
jokt
911
procedure
With the patient under gtmeral anesthesia, an arthrotomy of the right temporomandibular joint, including removal of t:re loose body and a high condylectomy, was performed from a preauricular incision. A Silastic implant was positioned in the joint space in the region occupied by tho resected condylar head. This implant was placed to prevent the possible development of ankylosis of the joint. The wound was then closed in layers in the usual manner. The patient tolerated the procedure extremely well, and postoperative recovery was uneventful. On the first postoperativl : day the patient was taking a full liquid diet without difficulty and had only moderate pain or discomfort. There was minimal edema associated with the site of the preauricular incision. On the third postoperative day sutures were removed from the preauricular incision, and the wound was observed to be healing well. At this time the patient was placed on a dental soft diet and started on a regimen of exercise to increase the maximum range of open’ng of the jaws. The patient continued to progress well without any marked degree of pain cmrdiscomfort, and by the fifth postoperative day she was able to open the jaws 2.5 cm., measured at the incisal edges of the anterior teeth. The patient was continued on an exercise routine and within 3 weeks was handling a regular diet well. She was discharged from the hospital on Aug. 19, 1966, to return home. Histopathology
Three specimens (HPS-490566 ABC) were submitted for histologic study and diagnosis. Specimen A, the loose body in the joint, measured 12 by 8 1)~ 5 mm., and gross examination revealed the specimen to be consistent with cartilage tissue. Specimen B, labeled “meniscus,” consisted of multiple fragments of tissue compatible with cartilage and fibrous tissue. Specimen B had a total volume of 3 ml. Specimen C was an irregular fragment of bone (condylar head) with a flat surface denoting that it had been surgically removed. This specimen measured 12 by G by 6 m:n. Sections of specimens A and B (loose body and meniscusj consisted primarily of sheets and clumps of actively grooving cartilage which were surrounded by dense hyalinized connective tissues (Fig. 3). Scattered remnants of synovial tissue were evident within the thickened connective tissue (Fig. 4). In many areas, there were transitional zones in which the connective tissue cells were plump and undergoing cartilaginous metaplasia (Fig. 5). The lesion was somewhat disturbing morphologically, in that the majority of cartilage cells were binucleate and multinucleate with hyperchromatic nuclei (Figs. 3 and 6). This pleomorphism is to be interpreted as actively growing cartilage and not as an index of malignant growth. Discrete areas of calcification and ossification were evident throughout the specimen (Fig. 7). Sections of specimen C consisted of relatively sclerotic bone tissue with a normal-appearing mature cartilaginous cap.
DISCUSSION
Although reports of chondrosarcoms arising from areas of synovial chondromatosis have been reported, we have been able to locate only one reported case of chondrosarcoma which developed from a previously radiographically and histologically diagnosed case of synovial chondromatosis.” It is postulated that in the other reported (:ases the lesions were actually chondrosarcoma from the start. Because of the morphology of the cells in chondromatosis, this condition could understandably be misdiagnosed as chondrosarcoma if one used only the histologic criteria. Therefore, a critical evaluation of the clinical and mdiographic evidence must be performed in arriving at a definite diagnosis. In this particular case, the lesions were evident for well over 18 months with no additional large appreciable areas of “Iytic” or “bIastic” degeneration in the area of the temporomandibular joint which, if present., would have been interpreted as a malignant, rapidly growing tumor. The preoperative diagnosis was one of benign degenerative arthritis. Only upon histologic examination of the tissue
912
Xchulte
08, O.M. & O.P. leic mher, 1 968
and Rhwns
Fig. 5
Fig.
Fig. cartilage. Fig. Fig. areas of
6
5. High-power
view
showing
metaplastio
transformation
of synovisl
remnants
into
6. High-power 7. High-power calcification.
view of typical cartilage cells of surgically removed specimens. view of loose body demonstrating pleomorphic cartilage cells and
Volume 28 Number 6
SynoGal
chondromatosis
of temporomandibular
joint
913
did the suspicion of chondrosarcoma arise, and only after extensive evaluation of the histologic features, behavior pattern of the tumor, subjective symptoms, and radiographs was the diagnosis of synovial chondromatosis established. The postoperative follow-up has been difficult, as the patient has moved to another geographic area. A personal letter confirms that she is free of symptoms at the present time and has as :Cunctional a temporomandibular joint as at any time in her life. SUMMARY
A single case of synovial chondromatosis has been presented. The disease entity is rare, when all articulating joints are considered, but the involvement of the temporomandibular joint is practically nonexistent. Although synovial chondromatosis is a benign process, the bizarre morphologic appearance of the tissue cells presents a difficult diagnostic problem in distinguishing between chondromatosis and chondrosarcoma. Management of the patient is, indeed, less dramatic and traumatic in the case of chondromatosis than when an erroneous diagnosis of chondrosarcoma is made. It is essential that radiologic, histologic, and clinical expressions be considered before a final diagnosis is made and surgical treatment is instigated. REFERENCES
1. Luck, V. C.: Bone and Joint Diseases, Springfield, Ill., 1950, Charles C Thomas, Publisher, pp. 576-579. 2. Jaffe, H. L. : Tumors and Tumorous Conditions of the Bones and Joints, Philadelphia, 1961, Lea & Febiger, pp. Em58-567. 3. Aegerter, E., and Eirkpa.trick J.: Orthopedic Diseases, ed. 2, Philadelphia, 1963, W. B. Saunders Company, p. 767. 4. Murphy, F. P., Dahlin, .D. C., and Sullivan, C. R.: Articular Synovial Chondromatosis, J. Bone & Joint Surg. 444.: 77-86, 19632. 5. Mullins, F., Berard, C. -W., and Eisenberg, 5. H.: Chondrosarcoma Following Synovial Chondromatosis, Cancer X3: 1180-1188, 1965.