10.59987/ads/2015.3-4.91-95
Aim. Synovial Chondromatosis (SC) is a rare, benign non neoplastic arthopathy characterized by the metaplastic development of cartilaginous nodules within the synovial membrane. In only 3% of all cases does it affect the temporomandibular joint (TMJ) and cases that arise from the lower compartment are rarely found in literature. The aim of this paper is to report a new case of SC of the inferior TMJ compartment with the description of the clinical, therapeutic and histopathological findings.
Case report. This article presents a 68-year-old woman with preauricular swelling on the right side, pain, crepitus and limited joint motion. This patient was evaluated by preoperative clinical manifestation, CT scan and MR images. Both showed multiple, calcified loose bodies in the inferior compartment. Based on these images as well as the patient’s signs and symptoms, a surgical intervention was performed. A good functional recovery with no signs of recurrence at 36 months of follow up was obtained.
Conclusion. Among cases of synovial chondromatosis in literature, only twelve originating in the lower compartment have been reported, this one included. In all the cases treated for SC in the lower compartment, both in literature and in our case report, surgical treatment led to healing.
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A rare case of synovial chondromatosis of the inferior TMJ compartment. Diagnosis and treatment aspect
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A rare case of synovial chondromatosis of the inferior TMJ compartment. Diagnosis and treatment aspect
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                                                                                                Review article



    A rare case of synovial chondromatosis
    of the inferior TMJ compartment.
    Diagnosis and treatment aspect




                                                                                                 i
                                                                                               al
                                                                                            on
    Davide Sozzi, MD1                                     In all the cases treated for SC in the lower com-
    Gabriele Bocchialini, MD1                             partment, both in literature and in our case report,
    Giorgio Novelli, MD1                                  surgical treatment led to healing.
    Maria Gabriella Valente, MD2
    Francesca Moltrasio, MD2                              Key words: inferior compartment, synovial chon-




                                                                                   zi
    Alberto Bozzetti, MD1                                 dromatosis, temporomandibular joint.




                                                                      na
    1 Maxillofacial O.U.S.Gerardo Hospital Monza, De-     Introduction
      partment of Medicine and Surgery, School of Medi-
      cine, University of Milano-Bicocca, Milan, Italy    Synovial chondromatosis (SC) is an uncommon, be-
    2 Pathology O.U. S.Gerardo Hospital Monza, Italy      nign lesion characterized by formation of small, multi-
                                                          ple, metaplastic nodules of cartilage generally within




                                                            er
                                                          the joint space.
                                                          SC usually affects large synovial joints such as the
    Corresponding author:                                 elbow, knee, wrist and hip. 3% of cases occurs in the



                                                      t
    Davide Sozzi                                          temporomandibular joint (TMJ) (1).
    Maxillofacial O.U.S.Gerardo Hospital Monza, Depart-
                                                   In
    ment of Medicine and Surgery, School of Medicine,
                                                          According to the literature more than 200 cases of SC
                                                          of the TMJ have been described (2) and the majority
    University of Milano-Bicocca                          of these arises in the upper compartment with various
    Via Cadore, 48                                        degrees of extension (3-7). This type of condition can
    20090 Monza, Italy                                    be locally aggressive and cases with intracranial ex-
                                               ni
    E-mail: davide.sozzi@unimib.it                        tension have been previously described (8-10).
                                                          International literature shows only eleven cases origi-
                                                          nating in the inferior compartment, before this case
                                    io

    Summary                                               (11-17).
                                                          This disease is also known as sinoval osteochondro-
    Aim. Synovial Chondromatosis (SC) is a rare, be-      matosis, synovial chondrometaplasy, synovial chon-
    nign non neoplastic arthopathy characterized by       drosis, synovial metaplasy, sinovialoma, and periar-
                           iz



    the metaplastic development of cartilaginous          ticular tenosynovial chondrometaplasy (3).
    nodules within the synovial membrane. In only 3%      In literature Milgram (18), Gerard (19) and Chen (17)
    of all cases does it affect the temporomandibular     have offered three different classifications.
          Ed




    joint (TMJ) and cases that arise from the lower       Milgram, in 1977, categorized the disease process in-
    compartment are rarely found in literature. The       to 3 distinct phases where we can find the relation-
    aim of this paper is to report a new case of SC of    ship between the cartilage foci and the synovium.
    the inferior TMJ compartment with the description     In phase I, metaplasia of the synovial intima occurs.
    of the clinical, therapeutic and histopathological    Active synovitis and nodule formation is present, but
    findings.                                             no calcifications can be identified.
IC




    Case report. This article presents a 68-year-old      In phase II, nodular synovitis and loose bodies are
    woman with preauricular swelling on the right         present in the joint. The loose bodies are primarily
    side, pain, crepitus and limited joint motion. This   still cartilaginous.
    patient was evaluated by preoperative clinical        In phase III, the loose bodies remain but the synovitis
C




    manifestation, CT scan and MR images. Both            has resolved. The loose bodies also have a tendency
    showed multiple, calcified loose bodies in the in-    to unite and calcify.
    ferior compartment. Based on these images as          The Gerard’s classification was completed in 1993
    well as the patient’s signs and symptoms, a surgi-    and was based on the synovial activity of the disease
                                                          and we can find 4 stages:
©




    cal intervention was performed. A good functional
    recovery with no signs of recurrence at 36 months     Stage 1: presence of cartilaginous of fibrocartilagi-
    of follow up was obtained.                            nous nodules with plenty of ground substance in the
    Conclusion. Among cases of synovial chondro-          synovium.
    matosis in literature, only twelve originating in     Stage 2: presence of a very thick synovium with nu-
    the lower compartment have been reported, this        merous small calcification or ossification cartilaginous
    one included.                                         nodules.

    Annali di Stomatologia 2015; VI (3-4): 91-95                                                               91
    D. Sozzi et al.


    Stage 3: presence of large and ossified nodules.
    Stage 4: the synovium is nearly normal or atrophic
    without any signs of metaplasia.
    Only in 2012 Chen et al. made an essential clinical-
    radiological classification according to the different




                                                                                                          i
    structures involved; they classified SC in the inferior




                                                                                                        al
    compartment of the TMJ into 3 stages.
    Stage 1 involves multiple loose bodies that are noted
    with expansion of the inferior compartment with no




                                                                                                     on
    bony erosion.
    Stage 2 involves multiple calcified nodules that are
    conglutinated to the condyle, the condyle is enlarged
    by pressure erosion but the disc is intact.




                                                                                           zi
    Stage 3 involves multiple calcified nodules congluti-              a
    nated to the condyle, the condyle in destroyed as a re-
    sult of a pressure erosion or by direct bony invasion of




                                                                            na
    the mass, the inferior surface of the disc is involved,
    and the lesion can’t be detached from the disc.
    The aim of this paper is to report a new case of syn-
    ovial chondromatosis of the inferior TMJ compart-
    ment with the description of the clinical, therapeutic




                                                                 er
                                                                  b
    and histopathological findings.
                                                               Figure 1 a-b. CT axial and coronal view showing calcifying
                                                               lesion and the normal contour of the right condyle (arrows).
    Case report


                                                         t
                                                      In
    A 68-year-old woman was referred to the O.U. of Maxil-
    lo-Facial Surgery, Monza S. Gerardo Hospital, Universi-
    ty of Milano-Bicocca School of Medicine with right sided
    preauricular swelling lasting 4 months, a limitation of
    mouth opening and pain aggravated by joint palpation
                                         ni
    and mastication. There was no history of trauma or oth-
    er event contributing to the onset of symptoms.
    The patient was edentulous and the maximal mouth
                                io


    opening was 20 mm with a deviation on the right side.
    The physical examination revealed that no facial
    nerve disfunction, hearing or facial sensation distur-
                        iz



    bances were detected.
    A computer tomographic scan (CTs) revealed the
    normal contour of the right condyle, no destruction of
          Ed




    the glenoid fossa or temporal bone on the same side,
    no widening of the joint space, no bony changes of
                                                               Figure 2. MRI sagittal view showing nodular images in the
    the skull base. It did reveal, however, several calcify-
                                                               inferior compartment with a little involvement in the superi-
    ing lesions in the right TMJ region (Fig. 1 a-b).          or compartment even (arrow).
    Magnetic resonance (MRI) showed multiple, calcified
    loose bodies in the inferior compartment; the disc
IC




    was irregular and patchy and its position was anterior
    to the condyle (Fig. 2).
    Because the CT and MRI imaging powerfully indicat-
    ed a benign lesion, no exploration biopsy was done.
C




    According to Chen’s classification (Stage I) a surgical
    intervention was performed under general anaesthe-
    sia with a right preauricular approach. The incision
    was followed by the exposure of the right temporo-
    mandibular region and cartilaginous nodules of vary-
©




    ing size were visible floating in the lower compart-
    ment (Fig. 3).
    All these cartilaginous fragments were removed to-
    gether with the affected synovium (Fig. 4).                Figure 3. Intraoperative view of the preauricular approach of
    Removed the nodules, the articular disc resumed its        the TMJ region during the removal of the loose bodies (White
    proper position.                                           arrow – external meatus, Black arrow – Zygomatic arch).

    92                                                                            Annali di Stomatologia 2015; VI (3-4): 91-95
    A rare case of synovial chondromatosis of the inferior TMJ compartment. Diagnosis and treatment aspect




                                                                                                           i
                                                                                                         al
                                                                                                      on
                                                                                             zi
                                                                  a




                                                                               na
    Figure 4. Macroscopic image of nodules of variable size
    from the lower compartment.


    After careful analysis and macroscopic description,




                                                                      er
    the surgical specimens were fixed in 4% buffered for-
    malin and paraffin-embedded.
    Subsequently, four-micron hematoxylin and eosin-stained
    sections of tumor were examined microscopically.
    Diagnostic criteria for SC included lobulated chon-


                                                           t
                                                        In
    droid proliferation with foci of hypercellularity and cal-
    cification within synovium and no underlying arthritis.
    Microscopically, our case presents nodules of hyaline
    cartilage lining by flattened synovial epithelium.            b
    These nodules were composed by clusters of chon-              Figure 5. (a) Photomicrograph of a typical loose body of
                                               ni
    drocytes and separated by a cartilaginous matrix. The         hypercellular cartilage covered with a layer of synovium
    chondrocytes showed nuclear hyperchromasia, en-               (EE, 40x). (b) High power view of lesion shows variable ar-
    largement and binuclear forms (Fig 5a-b).                     eas of atypia and hypercellularity (EE, 200x).
                                    io

    According to the radiological and histopathological as-
    pects, the SC in this patient was classified as a phase
    III in Milgram’s classification, as a stage II in Gerard’s
    classification and as a stage I in Chen’s classification.
                           iz



    One week after surgery the patient started physiothe-
    rapy for 45 days in order to improve and increase
    mouth opening and movement capability.
          Ed




    The CT was repeated at 6, 12 months and 24 months
    after the operation and confirmed the total removal of
    the tumor without any recurrence during the follow up
    period (Fig. 6 a-b). The patient was able to open her
    mouth to 35 mm with no deviation even though she
    reported slight pain during mandibular movement.
IC




    The follow up period is currently 36 months.

                                                                  a
    Discussion
C




    Synovial chondromatosis (SC) is a rare disease char-
    acterized by the formation of nodules of metaplastic
    cartilage under the surface of synovial membranes,
©




    joints, tendons and bursae.
    It was first described according to Ginaldi in 1558 by
    Ambrosio Pare (20); in 1764 Albrecht von Haller
    showed the first report in Elemental physiologiae cor-        b
    poris humani (21) and Axhausen in 1933 composed               Figure 6 a-b. CT axial and coronal view confirm the total
    the first detailed scientific work (22).                      removal of the tumor with no recurrence (24 months).



    Annali di Stomatologia 2015; VI (3-4): 91-95                                                                          93
    D. Sozzi et al.


    Afterwards, descriptions of this pathology have been           between the cartilage foci and the synovium is
    characterized by discrepancies of nomenclature.                placed, while Gerard’s classification (19) is based on
    The aetiology of SC is not clearly recognized but the          the synovial activity of the disease where 4 stages
    presence of clonal chromosomal aberrations sug-                can be recognized.
    gests the neoplastic nature of this condition (23).            An aggressive surgical treatment in the early stages




                                                                                                                i
    Cases are divided into two categories: primary SC,             is recommended both in Milgram’s and Gerard’s clas-




                                                                                                              al
    without specific aetiological factors that represent an        sification.
    active cartilaginous metaplasy originating in the syn-         The most recent clinical-radiological classification
    ovial membrane, and secondary SC related to previ-             was made by Chen et al. according to the different




                                                                                                           on
    ous trauma, repetitive microtrauma and degenerative            structures involved. They classified SC in the inferior
    arthritis or other arthropathies where a less cellular         compartment of the TMJ into 3 stages (17).
    atypia was found (24).                                         As above mentioned, different stages point out that
    Primary SC is deemed more aggressive (3).                      different structure are engaged; consequently differ-
                                                                   ent stages was supposed to have different principles




                                                                                                 zi
    The most common signs and symptoms that we
    found in the SC of the inferior compartment of the             of management (5).
    TMJ were: pain, swelling, limited mouth opening, joint         Guidelines for surgical treatment in these patients now
                                                                   derive from this latest and highly important classifica-




                                                                                 na
    crepitus and malocclusion. These symptoms were
    nonspecific.                                                   tion; so that in the first stage we can only remove both
    The differential diagnosis includes chondrosarcoma, os-        the loose bodies and the affected synovium, in the sec-
    teoarthritis, rheumatoid arthritis, avascular necrosis (25),   ond stage we could perform condilectomy, in the third
    osteochondrytis, intracapsular condylar fractures (24).        stage we could perform a condilectomy and discectomy.




                                                                     er
    The type of imaging techniques used for diagnosis              The reconstruction in the stage two and three could
    and surgical planning were CT scan and MRI.                    be made with costocondral graft or/and a pedicled
    CT scan findings consist in: multiple, calcified loose         deep temporal fascia fat flap (17).
    bodies in the joint space, widening of the joint space,        As previously revealed in our case report we found
    erosion of the glenoid fossa, bony changes of the

                                                            t      multiple, calcified loose bodies in the inferior com-
    skull base and condyle head (26, 27).
                                                         In        partment with a normal contour of the right condyle.
    The MRI found: intra-articular loose bodies present            Thus the removal of the loose bodies and affected
    as small areas of low signal intensity, massive expan-         synovium is generally enough, as Chen’s classifica-
    sion of the joint capsule, fluid accumulation within the       tion state; according to the radiological and clinical
    joint space and an irregular articular disc (27).              findings we perform this type of treatment in our pa-
                                            ni
    The histopathological aspects are represented macro-           tient with a preauricular approach.
    scopically by cartilaginous bodies that remain confined        No recurrence has been observed in the current 36-
    to the synovial membrane that may present as diffuse-          month-follow up period.
                                  io


    ly thickened with a cobblestone appearance or may              Open surgery has always been proposed as the ther-
    break off and protrude into the joint cavity as loose          apy of choice even though some Authors support a
    bodies. They may be large or small, cartilaginous, os-         less invasive procedure such as arthroscopy. This lat-
                         iz



    seous or both, ranging in number from a few to hun-            ter option is very difficult to carry out due to the posi-
    dreds. Microscopically there are several histopatholog-        tion of the disease (2).
    ical differences between primary and secondary forms
          Ed




    of SC (28, 29).
    Primary SC presents alterations such as crowding               Conclusion
    and irregularity of the cell nests, varying degrees of
    atypia of chondrocytes with plumped and binucleated            The case that we reported is the twelfth described in
    elements, which do not necessarily indicate a malig-           the literature. The treatment performed by us, after a
    nant transformation.                                           thorough evaluation of the literature, has proved ef-
IC




    Although the same increased cellularity occurs in              fective with low morbidity.
    secondary SC, it is uniformly distributed with more or-        In all the cases treated for SC in the lower compart-
    ganized cellular growth; chondrocytes also appear              ment, both in literature and in our case report, no re-
    regular and homogeneous.                                       currence has occurred so far.
C




    In addition, a further difference lies in the histological
    pattern of calcification: the primary form is patchy and
    diffuse whereas the secondary form is zonal with               References
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    A distinctive trait of secondary SC is the presence of nor-     1. Hohlweg-Majer B, Metzger MC, Bohm J, et al. Advanced
©




                                                                       imaging findings and computer-assisted surgery of suspected
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IC
C
©




    Annali di Stomatologia 2015; VI (3-4): 91-95                                                                                          95
