79th publication jmos- 2 nd name

CLOVE Dental & OMNI Hospitals
CLOVE Dental & OMNI HospitalsConsultant CranoFacial Surgeon & CEO https://www.writingandpublications.com/index.php em CLOVE Dental & OMNI Hospitals

Temporomandibular Joint Synovial Chondromatosis Posing as Diagnostic Dilemma: A Case Report J. Maxillofac. Oral Surg. (Oct–Dec 2019) 18(4):543–546.

CASE REPORT
Temporomandibular Joint Synovial Chondromatosis Posing
as Diagnostic Dilemma: A Case Report
Philip Mathew1 • Rahul V. C. Tiwari1 • Nikhil O. Govindan2
Received: 20 February 2018 / Accepted: 11 January 2019
Ó The Association of Oral and Maxillofacial Surgeons of India 2019
Introduction
Synovial Chondromatosis is a chronic process and a benign
tumor-like disorder which forms a loose cartilaginous
nodule in the synovial membrane. These nodules can be in
different shape and form, pedunculated, single or multiple
in number, free or attached to joint space, can be detached
from synovial membrane and also process a chance of
calcification. It was first described by Auhausen in 1933 in
the literature. Patients usually report with a history of
difficulty in mouth opening gradually with time and pain
with mild swelling on the preauricular area when involving
temporomandibular joint. The lesion hinders the normal
movements of temporomandibular joint. It is commonly
reported in the long bones [1]. In temporomandibular joint,
both unilateral and bilateral cases are reported with more
gravity to right side in unilateral cases [2]. Male/female
ratio is 1:1.5 in occurrence [2, 3]. It mainly occurs at the
age of 30–50 [3]. It has an idiopathic etiology, but the
literature reports joint disorders or trauma [4]. In cases of
severe reduction of joint movement, deviation can also be a
clinical feature toward unaffected side. Lesion is not
associated with pathology as reported till now. Clinically,
the examination has various differential diagnoses, but
magnetic resonance imaging or temporomandibular joint
tomography is helpful. Histopathology confirms the diag-
nosis. Removal of lesion is the treatment of choice.
Case Report
We report a case of 36-year-old male who was referred
from department of ENT as the patient was having a his-
tory of ear pain. Ultrasonography elicited a heterogeneous
nodule of 12 mm 9 19 mm 9 23 mm located posterior to
the posterior capsule of parotid gland closely attributing the
neck of mandibular condyle without cortical erosion. He
also complains about having pain in the temporo-
mandibular joint region of left side for 2 weeks and gives
history of difficulty in mouth opening and gradual reduc-
tion for 9 years which is idiopathic in origin. On extra oral
clinical examination, mild swelling was present on preau-
ricular area on the left temporomandibular joint region.
Tenderness was present on forceful wide mouth opening.
Jaw deviation was absent. Intraoral dental occlusion was
stable. Mouth opening was 33 mm. Clinical diagnosis was
temporomandibular joint disorder. Further radiographic
evaluation was performed by taking OPG which does not
give significant diagnosis. So magnetic resonance imaging
was advised which reported a small heterogeneous lesion
measuring 21 mm anteroposteriorly, 18 mm craniocau-
dally and 17 mm transverse along the margins of tem-
poromandibular joint surrounding the condyle and neck of
hemimandible involving parotid space (Fig. 1). The lesion
is of low-to-intermediate signal on T1W1, hypo intense,
somewhat heterogeneous in T2W1 and heterogeneously
& Philip Mathew
philip2mathew@gmail.com
Rahul V. C. Tiwari
drrahulvctiwari@gmail.com
Nikhil O. Govindan
drnikhilo@gmail.com
1
Department of Oral and Maxillofacial Surgery and Dentistry,
Jubilee Mission Medical College Hospital and Research
Institute, Thrissur, Kerala 680005, India
2
Department of Oral and Maxillofacial surgery, Sree Anjaneya
Institute of Dental Sciences, Modakkalloor, Calicut, Kerala,
India
123
J. Maxillofac. Oral Surg.
https://doi.org/10.1007/s12663-019-01186-0
enhancing. Mucosal thickening was present in bilateral
ethmoid and maxillary sinuses. Possibility of calcium
pyrophosphate dihydrate deposition disease and topha-
ceous/tumoral form was suspected, and differential diag-
nosis was given as temporomandibular joint synovial
chondromatosis as no remodeling or mass effect on con-
dyle with intracranial involvement was noted. Plain helical
computed tomography scan of left temporomandibular
joint showed a soft tissue density lesion (40–55 Hounsfield
Unit) in close relation to condyle of mandible and posterior
aspect of ramus and left masseter measuring 20 mm
craniocaudally, 14 mm transverse and 20 mm anteropos-
teriorly (Fig. 2). Minimal scalloping was noted at the lat-
eral aspect of condyle. There was no extension into
temporomandibular joint or infratemporal fossa. Lateral
and inferior part of the lesion is closely abutting superficial
lobe of parotid gland. After a thorough evaluation of
clinical and radiographic diagnosis, excision of lesion was
planned under general anesthesia. A preauricular approach
was taken, and joint space was exposed (Fig. 3). Lesion
was separated from the surrounding vital borders and
margins, and excised. Excised tissue was sent for
histopathologic evaluation, showing macroscopically a
gray-brown nodule of irregular surface with cut sections
showing small yellowish white bits. Esthetic closure was
performed. Histopathology (9 40) reported a small meta-
plastic cartilaginous nodule of varying size with calcifica-
tion and also synovial tissue along with nodules of
cartilage, normal chondrocytes are noted with occasional
foci of calcification which confirmed the clinicoradio-
graphic diagnosis. Postoperatively, patient had an adequate
mouth opening with minimal scar on the preauricular
incision area and no pain in normal temporomandibular
joint movements (Fig. 4).
Discussion
Synovial chondrometaplasia, articular chondromatosis,
osteochondromatosis, synovial osteochondromatosis,
chondrocalcinosis articularis, tenosynovial chondromato-
sis, joint chondromatosis) metaplastic change of mes-
enchymal cells in the joint synovial tissue into cartilage [4].
Tgf-1 involvement in chondrogenesis abnormalities has
been reported on chromosome 6 Milgram’s phases i) active
synovial disease, no loose bodies ii) active synovial pro-
liferation, free loose bodies iii) multiple loose bodies, no
synovial disease [5]. Temporomandibular joint synovial
chondromatosis is classified histopathologically and radi-
ologically. Histologically, synovial chondromatosis may be
divided into three stages of development: (1) metaplasia
found in the synovial membrane without the presence of
detached particles, (2) metaplasia found in the synovial
membrane with the presence of detached particles and (3)
only detached particles, which may vary in size from less
than 1 mm to greater than 10 mm [6]. Noyek and
coworkers (1) pointed out radiologic features of synovial
chondromatosis in the TMJ, namely (1) widening of the
joint space, (2) limitation of motion, (3) irregularity of the
joint surface, (4) presence of calcified loose bodies (carti-
lage) and (5) sclerosis or hyperostosis (overgrowth) of the
glenoid fossa and mandibular condyle [7]. Differential
diagnoses of this lesion are sarcomatous degeneration
(rare), synovial chondrosarcoma, loose bodies,
Fig. 1 Magnetic resonance imaging-axial section
Fig. 2 Temporomandibular joint computed tomography
J. Maxillofac. Oral Surg.
123
osteochondritis dissecans, neurotrophic arthritis, tubercu-
lous arthritis, osteochondral fibrosis, osteonecrosis, periar-
ticular pseudogout, amyloid arthropathy (when disease is
active), pigmented villonodular synovitis (when no calci-
fications present). Familial synovial chondromatosis has
been reported in three men of the same family (x-linked) in
two generations. Multifocal synovial chondromatosis has
been reported with bilateral TMJ occurrence, simultaneous
synovial chondromatosis and pigmented villonodular syn-
ovitis (PVNS). Simultaneous involvement of both diseases
in one joint has been reported TMJ: iatrogenic chondro-
matosis has been reported 8 months after the autologous
chondrocyte implantation, and secondary synovial chon-
dromatosis occurrence within a bursal sac overlying an
osteochondroma has been reported which mimics a chon-
drosarcoma radiologically [8]. In the animal model, it is
found in dog which had shoulder involvement has been
reported as a cause of lameness [9]. The peculiar gross and
histopathologic features are nodules of cartilage adjacent to
synovium, often connected only by a stalk. Intra-articular
cartilaginous loose bodies ‘‘snow storm knee’’ have been
described with hundreds of loose bodies without synovitis.
Central area of loose body loses nutrition source, under-
goes necrosis and calcifies. Synovial metaplasia to cartilage
substantiated by lack of immunohistochemical detection of
the Ki-67 protein (proliferation marker) [10].
Conclusion
Temporomandibular joint synovial chondromatosis is
rarely reported worldwide. To the best of our knowledge in
the published literature, it is a rare case reported in India.
As the lesion is benign and slow growing, it also creates
functional disturbance and psychological imbalance to
patient. Clinical examination alone is not self-satisfactory
to come with a diagnosis regarding such cases, but also
radiology and histopathology are equally important.
References
1. Fuller E, Bharatha A, Yeung R, Kassel EE, Aviv RI, Howard P,
Symons SP (2011) Case of the month #166: synovial chondro-
matosis of the temporal mandibular joint. Can Assoc Radiol J
62(2):151–153
2. Guijarro-Martı´nez R, Torres MP, Mateo MM, Garcı´a IS, Alba
LM, Gimilio MEI et al (2011) Bilateral synovial chondromatosis
of the temporomandibular joint. J Cranio Maxillofac Facial Surg
39:261–265
3. Bonatti BDS, Patrocinio LG, Costa SAA, Costa JMC, Patrocinio
JA (2008) Condromatose sinovial de articulac¸a˜o teˆmporo-
mandibular. Rev Bras Otorrinolaringol 74(3):480
4. Munk PL, Helms CA (1989) Temporomandibular joint synovial
chondromatosis: CT manifestations. J Can Assoc Radiol
40:274–276
5. Nitzan DW, Marmary Y et al (1991) The diagnostic value of
computed tomography in temporomandibular joint synovial
chondromatosis. Comput Med Imaging Graph 15:53–56
6. Doran EA, Vogler JB, Angelillo JC (1989) Synovial chondro-
matosis of the temporo-mandibular joint diagnosed by magnetic
resonance imaging. J Oral Maxillofac Surg 47:411–413
Fig. 3 Exposure and excision
of lesion
Fig. 4 Histopathologic slide of lesion
J. Maxillofac. Oral Surg.
123
7. Noyek AM, Holgate RC, Fireman SM, Rosen P, Pritzker KPH
(1977) The radiological findings in synovial chondromatosis
(chondrometaplasia) of the temporomandibular joint. J Otolaryn-
gol 6:45–48
8. Murphey MD, Vidal JA, Fanburg-Smith JC, Gajewski DA (2007)
Imaging of synovial chondromatosis with radiologic-pathologic
correlation. Radiographics 27:1465–1488
9. Kunkel KA, Rochat MC (2008) A review of lameness
attributable to the shoulder in the dog: part two. J Am Anim Hosp
Assoc 44:163–170
10. Shah SB, Ramanojam S, Gadre PK, Gadre KS (2011) Synovial
chondromatosis of temporomandibular joint. Journey through 25
decades and a case report. J Oral Maxillofac Surg 69:2795–2814
Publisher’s Note Springer Nature remains neutral with regard to
jurisdictional claims in published maps and institutional affiliations.
J. Maxillofac. Oral Surg.
123

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79th publication jmos- 2 nd name

  • 1. CASE REPORT Temporomandibular Joint Synovial Chondromatosis Posing as Diagnostic Dilemma: A Case Report Philip Mathew1 • Rahul V. C. Tiwari1 • Nikhil O. Govindan2 Received: 20 February 2018 / Accepted: 11 January 2019 Ó The Association of Oral and Maxillofacial Surgeons of India 2019 Introduction Synovial Chondromatosis is a chronic process and a benign tumor-like disorder which forms a loose cartilaginous nodule in the synovial membrane. These nodules can be in different shape and form, pedunculated, single or multiple in number, free or attached to joint space, can be detached from synovial membrane and also process a chance of calcification. It was first described by Auhausen in 1933 in the literature. Patients usually report with a history of difficulty in mouth opening gradually with time and pain with mild swelling on the preauricular area when involving temporomandibular joint. The lesion hinders the normal movements of temporomandibular joint. It is commonly reported in the long bones [1]. In temporomandibular joint, both unilateral and bilateral cases are reported with more gravity to right side in unilateral cases [2]. Male/female ratio is 1:1.5 in occurrence [2, 3]. It mainly occurs at the age of 30–50 [3]. It has an idiopathic etiology, but the literature reports joint disorders or trauma [4]. In cases of severe reduction of joint movement, deviation can also be a clinical feature toward unaffected side. Lesion is not associated with pathology as reported till now. Clinically, the examination has various differential diagnoses, but magnetic resonance imaging or temporomandibular joint tomography is helpful. Histopathology confirms the diag- nosis. Removal of lesion is the treatment of choice. Case Report We report a case of 36-year-old male who was referred from department of ENT as the patient was having a his- tory of ear pain. Ultrasonography elicited a heterogeneous nodule of 12 mm 9 19 mm 9 23 mm located posterior to the posterior capsule of parotid gland closely attributing the neck of mandibular condyle without cortical erosion. He also complains about having pain in the temporo- mandibular joint region of left side for 2 weeks and gives history of difficulty in mouth opening and gradual reduc- tion for 9 years which is idiopathic in origin. On extra oral clinical examination, mild swelling was present on preau- ricular area on the left temporomandibular joint region. Tenderness was present on forceful wide mouth opening. Jaw deviation was absent. Intraoral dental occlusion was stable. Mouth opening was 33 mm. Clinical diagnosis was temporomandibular joint disorder. Further radiographic evaluation was performed by taking OPG which does not give significant diagnosis. So magnetic resonance imaging was advised which reported a small heterogeneous lesion measuring 21 mm anteroposteriorly, 18 mm craniocau- dally and 17 mm transverse along the margins of tem- poromandibular joint surrounding the condyle and neck of hemimandible involving parotid space (Fig. 1). The lesion is of low-to-intermediate signal on T1W1, hypo intense, somewhat heterogeneous in T2W1 and heterogeneously & Philip Mathew philip2mathew@gmail.com Rahul V. C. Tiwari drrahulvctiwari@gmail.com Nikhil O. Govindan drnikhilo@gmail.com 1 Department of Oral and Maxillofacial Surgery and Dentistry, Jubilee Mission Medical College Hospital and Research Institute, Thrissur, Kerala 680005, India 2 Department of Oral and Maxillofacial surgery, Sree Anjaneya Institute of Dental Sciences, Modakkalloor, Calicut, Kerala, India 123 J. Maxillofac. Oral Surg. https://doi.org/10.1007/s12663-019-01186-0
  • 2. enhancing. Mucosal thickening was present in bilateral ethmoid and maxillary sinuses. Possibility of calcium pyrophosphate dihydrate deposition disease and topha- ceous/tumoral form was suspected, and differential diag- nosis was given as temporomandibular joint synovial chondromatosis as no remodeling or mass effect on con- dyle with intracranial involvement was noted. Plain helical computed tomography scan of left temporomandibular joint showed a soft tissue density lesion (40–55 Hounsfield Unit) in close relation to condyle of mandible and posterior aspect of ramus and left masseter measuring 20 mm craniocaudally, 14 mm transverse and 20 mm anteropos- teriorly (Fig. 2). Minimal scalloping was noted at the lat- eral aspect of condyle. There was no extension into temporomandibular joint or infratemporal fossa. Lateral and inferior part of the lesion is closely abutting superficial lobe of parotid gland. After a thorough evaluation of clinical and radiographic diagnosis, excision of lesion was planned under general anesthesia. A preauricular approach was taken, and joint space was exposed (Fig. 3). Lesion was separated from the surrounding vital borders and margins, and excised. Excised tissue was sent for histopathologic evaluation, showing macroscopically a gray-brown nodule of irregular surface with cut sections showing small yellowish white bits. Esthetic closure was performed. Histopathology (9 40) reported a small meta- plastic cartilaginous nodule of varying size with calcifica- tion and also synovial tissue along with nodules of cartilage, normal chondrocytes are noted with occasional foci of calcification which confirmed the clinicoradio- graphic diagnosis. Postoperatively, patient had an adequate mouth opening with minimal scar on the preauricular incision area and no pain in normal temporomandibular joint movements (Fig. 4). Discussion Synovial chondrometaplasia, articular chondromatosis, osteochondromatosis, synovial osteochondromatosis, chondrocalcinosis articularis, tenosynovial chondromato- sis, joint chondromatosis) metaplastic change of mes- enchymal cells in the joint synovial tissue into cartilage [4]. Tgf-1 involvement in chondrogenesis abnormalities has been reported on chromosome 6 Milgram’s phases i) active synovial disease, no loose bodies ii) active synovial pro- liferation, free loose bodies iii) multiple loose bodies, no synovial disease [5]. Temporomandibular joint synovial chondromatosis is classified histopathologically and radi- ologically. Histologically, synovial chondromatosis may be divided into three stages of development: (1) metaplasia found in the synovial membrane without the presence of detached particles, (2) metaplasia found in the synovial membrane with the presence of detached particles and (3) only detached particles, which may vary in size from less than 1 mm to greater than 10 mm [6]. Noyek and coworkers (1) pointed out radiologic features of synovial chondromatosis in the TMJ, namely (1) widening of the joint space, (2) limitation of motion, (3) irregularity of the joint surface, (4) presence of calcified loose bodies (carti- lage) and (5) sclerosis or hyperostosis (overgrowth) of the glenoid fossa and mandibular condyle [7]. Differential diagnoses of this lesion are sarcomatous degeneration (rare), synovial chondrosarcoma, loose bodies, Fig. 1 Magnetic resonance imaging-axial section Fig. 2 Temporomandibular joint computed tomography J. Maxillofac. Oral Surg. 123
  • 3. osteochondritis dissecans, neurotrophic arthritis, tubercu- lous arthritis, osteochondral fibrosis, osteonecrosis, periar- ticular pseudogout, amyloid arthropathy (when disease is active), pigmented villonodular synovitis (when no calci- fications present). Familial synovial chondromatosis has been reported in three men of the same family (x-linked) in two generations. Multifocal synovial chondromatosis has been reported with bilateral TMJ occurrence, simultaneous synovial chondromatosis and pigmented villonodular syn- ovitis (PVNS). Simultaneous involvement of both diseases in one joint has been reported TMJ: iatrogenic chondro- matosis has been reported 8 months after the autologous chondrocyte implantation, and secondary synovial chon- dromatosis occurrence within a bursal sac overlying an osteochondroma has been reported which mimics a chon- drosarcoma radiologically [8]. In the animal model, it is found in dog which had shoulder involvement has been reported as a cause of lameness [9]. The peculiar gross and histopathologic features are nodules of cartilage adjacent to synovium, often connected only by a stalk. Intra-articular cartilaginous loose bodies ‘‘snow storm knee’’ have been described with hundreds of loose bodies without synovitis. Central area of loose body loses nutrition source, under- goes necrosis and calcifies. Synovial metaplasia to cartilage substantiated by lack of immunohistochemical detection of the Ki-67 protein (proliferation marker) [10]. Conclusion Temporomandibular joint synovial chondromatosis is rarely reported worldwide. To the best of our knowledge in the published literature, it is a rare case reported in India. As the lesion is benign and slow growing, it also creates functional disturbance and psychological imbalance to patient. Clinical examination alone is not self-satisfactory to come with a diagnosis regarding such cases, but also radiology and histopathology are equally important. References 1. Fuller E, Bharatha A, Yeung R, Kassel EE, Aviv RI, Howard P, Symons SP (2011) Case of the month #166: synovial chondro- matosis of the temporal mandibular joint. Can Assoc Radiol J 62(2):151–153 2. Guijarro-Martı´nez R, Torres MP, Mateo MM, Garcı´a IS, Alba LM, Gimilio MEI et al (2011) Bilateral synovial chondromatosis of the temporomandibular joint. J Cranio Maxillofac Facial Surg 39:261–265 3. Bonatti BDS, Patrocinio LG, Costa SAA, Costa JMC, Patrocinio JA (2008) Condromatose sinovial de articulac¸a˜o teˆmporo- mandibular. Rev Bras Otorrinolaringol 74(3):480 4. Munk PL, Helms CA (1989) Temporomandibular joint synovial chondromatosis: CT manifestations. J Can Assoc Radiol 40:274–276 5. Nitzan DW, Marmary Y et al (1991) The diagnostic value of computed tomography in temporomandibular joint synovial chondromatosis. Comput Med Imaging Graph 15:53–56 6. Doran EA, Vogler JB, Angelillo JC (1989) Synovial chondro- matosis of the temporo-mandibular joint diagnosed by magnetic resonance imaging. J Oral Maxillofac Surg 47:411–413 Fig. 3 Exposure and excision of lesion Fig. 4 Histopathologic slide of lesion J. Maxillofac. Oral Surg. 123
  • 4. 7. Noyek AM, Holgate RC, Fireman SM, Rosen P, Pritzker KPH (1977) The radiological findings in synovial chondromatosis (chondrometaplasia) of the temporomandibular joint. J Otolaryn- gol 6:45–48 8. Murphey MD, Vidal JA, Fanburg-Smith JC, Gajewski DA (2007) Imaging of synovial chondromatosis with radiologic-pathologic correlation. Radiographics 27:1465–1488 9. Kunkel KA, Rochat MC (2008) A review of lameness attributable to the shoulder in the dog: part two. J Am Anim Hosp Assoc 44:163–170 10. Shah SB, Ramanojam S, Gadre PK, Gadre KS (2011) Synovial chondromatosis of temporomandibular joint. Journey through 25 decades and a case report. J Oral Maxillofac Surg 69:2795–2814 Publisher’s Note Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. J. Maxillofac. Oral Surg. 123