- Email: [email protected]
Severe Isolated Temporomandibular Joint Involvement in Juvenile Idiopathic Arthritis
1368
SEVERE ISOLATED TMJ INVOLVEMENT IN JIA
6. Pindborg JJ, Vedtofte P, Reibel J, et al: The calcifying epithelial odontogenic tumor. A review of recent literature and report of a case. APMIS Suppl 1991;23:152 7. Morimoto C, Tsujimoto M, Shimaoka S, et al: Ultrastructural localization of alkaline phosphatase in the calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol 56:409, 1983 8. Chomette G, Auriol M, Guilbert F: Tumeur épithéliale odontogéne calcifiée bifocale (Tumeur de Pindborg). Rev Stomatol Chir Maxillofac 85:329, 1984 9. Damm DD, White DK, Drummond JF, et al: Combined epithelial odontogenic tumor: Adenomatoid odontogenic tumor and calcifying epithelial odontogenic tumor. Oral Surg 55:487, 1983 10. Bingham RA, Adrian JC: Combined epithelial odontogenic tumor - Adenomatoid odontogenic tumor and calcifying epithelial odontogenic tumor: report of a case. J Oral Maxillofac Surg 44:574, 1986 11. Takeda Y, Kudo K: Adenomatoid odontogenic tumor associated with calcifying epithelial odontogenic tumor. Int J Oral Maxillofac Surg 15:469, 1986 12. Hicks MJ, Flaitz CM, Wong ME, et al: Clear cell variant of calcifying epithelial odontogenic tumor: Case report and review of the literature. Head Neck 16:272, 1994 13. Melrose RJ: Benign epithelial odontogenic tumors. Semin Diagn Pathol 16:271, 1999 14. Veness MJ, Morgan G, Collins AP, et al: Calcifying epithelial odontogénico (Pindborg) tumor with malignant transformation and metastatic spread. Head Neck 23:692, 2001
15. Cross JJ, Pilkington RJ, Antoun NM, et al: Value of computed tomography and magnetic resonance imaging in the treatment of a calcifying epithelial odontogenic (Pindborg) tumour. Br J Oral Maxillofac Surg 38:154, 2000 16. Slootweb PJ: Bone and cementum as stromal features in Pindborg tumor. J Oral Pathol Med 20:93, 1991 17. El-Labban NG: Cementum-like material in a case of Pindborg tumor. J Oral Pathol Med 19:166, 1990 18. Chomette G, Auriol M, Guilbert F: Histoenzymological and ultrastructural study of a bifocal calcifying epithelial odontogenic tumor. Characteristics of epithelial cells and histogénesis of amyloidlike material. Virchows Arch (Pathol Anat) 403:67, 1984 19. El-Labban NG, Lee KW, Kramer IRH: The duality of the cell population in a calcifying odontogenic tumor (CEOT). Histopathology 8:679, 1984 20. Ai-Ru L, Zhen L, Jian S: Calcifying epithelial odontogenic tumors: A clinicopathologic study of nine cases. J Oral Pathol 11:399, 1982 21. Maiorano E, Altini M, Favia G: Clear cell tumors of the salivary glands, jaws and oral mucosa. Semin Diagn Pathol 14:203, 1997 22. Veness MJ, Morgan G, Collins AP, et al: Calcifying epithelial odontogénico (Pindborg) tumor with malignant transformation and metastatic spread. Head Neck 23:692, 2001 23. Basu MK, Matthews JB, Sear AJ, et al: Calcifying epithelial odontogenic tumor: A case showing features of malignancy. J Oral Pathol 13:310, 1984
J Oral Maxillofac Surg 63:1368-1371, 2005
Severe Isolated Temporomandibular Joint Involvement in Juvenile Idiopathic Arthritis Paolo Scolozzi, MD, DMD,* Geraldine Bosson, MD, DMD,† and Bertrand Jaques, MD, DMD‡ First described by Meyer Diamant-Berger in 1892 and by Frederic Still in 1897, juvenile rheumatoid arthritis represents a heterogeneous group of chronic inflammatory arthritis that begins in childhood and is distinct from adult rheumatoid arthritis.1 In 1997, the International League of Associations for RheumatolReceived from the Department of Otolaryngology/Head and Neck Surgery, Division of Oral and Maxillofacial Surgery, Centre Hospitalier Universitaire Vaudois, Lausanne, Switzerland. *Chief Resident. †Resident. ‡Professor. Address correspondence and reprint requests to Dr Scolozzi: Department of Otolaryngology/Head and Neck Surgery, Division of Oral and Maxillofacial Surgery, Centre Hospitalier Universitaire Vaudois, 1011 Lausanne, Switzerland; e-mail: [email protected] © 2005 American Association of Oral and Maxillofacial Surgeons
0278-2391/05/6309-0018$30.00/0 doi:10.1016/j.joms.2005.05.300
ogy proposed the more universal term of “juvenile idiopathic arthritis” (JIA) and recognized 8 categories: systemic arthritis, oligoarthritis (arthritis affecting 1 to 4 joints in the first 6 months of the disease), extended oligoarthritis, polyarthritis (more than 4 joints in the first 6 months of disease with 2 subgroups, rheumatoid factor (positive or negative), enthesitis related arthritis (largely made up of HLA B-27 related disease), psoriatic arthritis, and “other arthritis.” Juvenile idiopathic arthritis refers to arthritis of unknown cause in 1 or more joints of at least 6 weeks’ duration occurring in children less than 16 years old.2 Juvenile idiopathic arthritis commonly afflicts the temporomandibular joint (TMJ) leading to the destruction of the condylar growth center and subsequent mandibular growth disturbances in severe cases.3-6 This can result in unpleasant facial deformity such as asymmetry, micro- and retrognathia, as well as secondary abnormal dental occlusion development. The ultimate and dramatic consequence of the joint destruc-
1369
SCOLOZZI, BOSSON, AND JAQUES
FIGURE 1. Coronal (A and C) and sagittal oblique (B and D) reconstructions from a thin-slice spiral computed tomography acquisition with bone algorithm through both TMJs shows bilateral extensive flattening of condyles and multiple cortical erosions of the glenoid fossa. Scolozzi, Bosson, and Jaques. Severe Isolated TMJ Involvement in JIA. J Oral Maxillofac Surg 2005.
tion will result in TMJ ankylosis and a definitive limitation of the bite.3-6 We are reporting the case of a 5-year-old girl who presented with isolated bilateral TMJ arthritis without any other articular involvement as exclusive manifestation of a JIA. To the best of our knowledge, only 1 similar case has been reported previously.7
Report of a Case In October 2003, a 5-year-old child was referred by her otolaryngologist for investigation of severe bilateral TMJ pain and trismus of 4 week’s duration. The patient was afebrile and otherwise in good health and extra- and intraoral examinations were normal. The function of the TMJ was evaluated bilaterally for calculation of the Dysfunction Index as described by Hel-
FIGURE 2. Gadolinium-enhanced T1-weighted images revealed a marked thickened inflammatory synovium (pannus) and abundant intra-articular effusion corresponding to synovitis (white arrow) (A-D). Note flattening of the disc otherwise in a normal position (black arrow) (B-D). Scolozzi, Bosson, and Jaques. Severe Isolated TMJ Involvement in JIA. J Oral Maxillofac Surg 2005.
1370 kimo.8 Both TMJs showed signs of severe dysfunction (Di III) with tenderness to palpation of the joint, pain during mouth opening, and limited mobility of the condyles. Mouth opening was restricted to 10 mm and protrusive as well as lateral movements were impossible. There were no spasms in the muscles of mastication and no joint sounds. The otoscopic examination, as well as the rest of the physical examination, was normal. Panoramic radiograph revealed flattening and important bilateral condylar destruction with a narrowed articular space. A complete chemical and microbiological screening revealed no abnormalities. Serological markers were negative for viral hepatitis B and C, Borrelia burgdorferi, Chlamydia psittaci and C. trachomatis, Mycoplasma pneumoniae, and HIV. Rheumatoid factor and antinuclear antibody were negative. The patient underwent a spiral computed tomography acquisition (1 mm slices, pitch 1, overlapping reconstructions every 0.5 mm, bone algorithm) as well as magnetic resonance (MR) examination of the TMJ (head coil, axial 3 mm T2 FSE sequences, axial and coronal 3 mm T1 SE images before and after injection of Gadolinium chelates). Computed tomography and MRI showed bilateral extensive decrease of the joint space as well as a marked flattening of both condyle and glenoid fossa with multiple cortical erosions (Figs 1 and 2). Gadolinium-enhanced T1-weighted images revealed a marked enhancing thickened inflammatory synovium (pannus) and abundant intra-articular effusion. The disc was thinned, but in a normal position at rest and during translation on both sides (Fig 2). In November 2003, given the difficulty in eating caused by the severe limitation of mouth opening as well as the negative response to the NSAID, we decided to perform a bilateral TMJ arthrocentesis with lavage. The procedure was performed under general anesthesia, and 1 mL of slightly hemorrhagic synovial fluid was obtained and the joints were lavaged with 100 mL of NaCl 0.9% using 2 large bore needles. Analysis of all the fluid obtained revealed 200 leucocytes/mm3 and no crystals. Cultures were sterile. The final diagnosis was that of a JIA isolated to the TMJ. The patient’s condition improved rapidly with total disappearance of the TMJ pain within the first 24 hours postoperatively. Physiotherapy with protrusion-opening jaw exercises was started 1 week after the procedure. At the 6-month followup, the patient was pain free and the maximum mouth opening achieved was 40 mm, protrusion movement was 6 mm, right and left lateral movements were 14 mm.
Discussion In the reported case, our patient presented with severe bilateral TMJ arthritis with effusion as well as advanced cortical destruction of both the condyle and the glenoid fossa. In children, 2 main diseases can produce such TMJ lesions: septic arthritis and JIA. Septic arthritis of the TMJ typically affects young children and follows a middle ear infection. Vestigial foramen and fissures between the TMJ and the middle ear, which usually close during the first 2 years of life, account for possible reciprocal spread of inflammation or infection between these 2 organs. Since the introduction of antibiotics, the incidence has been dramatically reduced.9 This diagnosis was ruled out in our patient by the cultures of the synovial liquid, which were sterile.
SEVERE ISOLATED TMJ INVOLVEMENT IN JIA
Although TMJ involvement in children with JIA is not uncommon, it rarely occurs at the beginning of the disease.1-7,10 Other joints such as the knee, hands and feet, hips, and sacroiliac joints are generally involved first. Its prevalence has been reported to be highest in polyarticular, early onset and antinuclear-positive arthritis.4 The most commonly reported symptoms are pain during function and at rest, morning stiffness, restricted maximal voluntary mouth opening, and TMJ crepitation. Patients with JIA of the TMJ can also exhibit functional impairments such as painful TMJ clicking and locking mimicking an internal derangement.5 The prevalence of reported radiologic TMJ abnormalities in children with JIA have varied from 29% to 66% in the literature, with cortical erosions and flattening of the condyle being the most common lesions found.4-6 Svensson et al5 showed that radiographic condylar lesions were significantly correlated with postnormal occlusion, restricted mouth opening, anterior open bite, and mandibular retrognathia. They did not find any significant correlation between condylar lesions and TMJ pain. The main concern in our patient was the isolated involvement of both TMJs without any other articular involvement during 6 months’ follow-up. In such specific cases where the clinical presentation is atypical and unusual, imaging (particularly MRI) is of value in detecting and evaluating intraarticular abnormalities.4-6 MRI is the most sensitive radiologic indicator of disease activity.6 In TMJ involvement, it is more sensitive than clinical evaluation, demonstrating inflammatory changes even in the absence of clinical symptoms.6 In our case, MRI provided the final diagnosis showing the characteristic lesions found in JIA, namely synovial hypertrophy (pannus), intra-articular effusion, and cartilaginous and subchondral bone erosions. Regarding the management of TMJ involvement in JIA, a general consensus concerning treatments does not exist, and they are often extrapolated from those used for other articulations, especially of the knee. Preliminary beneficial effects of simple TMJ arthrocentesis with lavage, without arthroscopy (as proposed by Nitzan et al11) have been reported in adults with rheumatoid arthritis, whereas reports in the pediatric population are lacking. The intra-articular lavage washes the inflamed synovial liquid, which in JIA patients contains different inflammatory proteins and lipid pain mediators.11,12 These proteins and lipids apparently act on nerve endings of the synovial membrane-provoking nociception. The final outcomes of this procedure are decreased pain, increased maximal mouth opening, and improved lateral movements.11,12 Arthroscopy alone or followed by synovectomy has been reported in the orthopedic literature especially for active knee synovitis, whereas only 1 case has
1371
GLIDDON ET AL
been reported for treatment of TMJ involvement in a patient with JIA.7 Intra-articular steroid injections in JIA have been attempted based on the experience with adults.13,14 Although it is a safe and rapidly effective treatment for synovitis, there are no data to confirm whether the disease course is modified in the long term and the literature lacks controlled studies. Its use remains anecdotal. Vallon et al14 found that the long-term development of symptoms and signs from the TMJ and the long-term progression of the joint destruction were low in patients previously treated with steroid intra-articular injections. Despite the good response of our patient after the arthrocentesis and lavage, the management of JIA remains an unsolved dilemma and a great challenge for the future, especially in limiting dramatic growth disturbances.
4.
5.
6. 7.
8.
9.
10. 11.
12.
References 1. Schneider R, Passo MH: Juvenile rheumatoid arthritis. Rheum Dis Clin North Am 28:503, 2002 2. Petty RE, Southwood TR, Baum J, et al: Revision of the proposed classification criteria for juvenile idiopathic arthritis: Durban, 1997. J Rheumatol 25:1991, 1998 3. Svensson B, Larsson A, Adell R: The mandibular condyle in juvenile chronic arthritis patients with mandibular hypoplasia:
13.
14.
A clinical and histological study. Int J Oral Maxillofac Surg 30:300, 2001 Pedersen TK, Jensen JJ, Melsen B, et al: Resorption of the temporomandibular condylar bone according to subtypes of juvenile chronic arthritis. J Rheumatol 28:2109, 2001 Svensson B, Adell R, Kopp S: Temporomandibular disorders in juvenile chronic arthritis patients. A clinical study. Swed Dent J 24:83, 2000 Johnson K, Gardner-Medwin J: Childhood arthritis: Classification and radiology. Clin Radiol 57:47, 2002 Martini G, Bacciliero U, Tregnaghi A, et al: Isolated temporomandibular synovitis as unique presentation of juvenile idiopathic arthritis. J Rheumatol 28:1689, 2001 Helkimo M: Studies on function and dysfunction of the masticatory system. II. Index for anamnestic and clinical dysfunction and occlusal state. Swed Dent J 67:101, 1974 Scolozzi P, Becker M, Richter M: Temporomandibular joint osteoarthritis: A cause of a serous otitis media? A case report. J Oral Maxillofac Surg 62:97, 2004 Kulas DT, Schanberg L: Juvenile idiopathic arthritis. Curr Opin Rheumatol 13:392, 2001 Nitzan DW, Dolwick MF, Martinez GA: Temporomandibular joint arthrocentesis: A simplified treatment for severe, limited mouth opening. J Oral Maxillofac Surg 49:1163, 1991 Trieger N, Hoffman CH, Rodriguez E: The effect of arthrocentesis of the temporomandibular joint in patients with rheumatoid arthritis. J Oral Maxillofac Surg 57:537, 1999 Cleary AG, Murphy HD, Davidson JE: Intra-articular corticosteroid injections in juvenile idiopathic arthritis. Arch Dis Child 88:192, 2003 Vallon D, Akerman S, Nilner M, et al: Long-term follow-up of intra-articular injections into the temporomandibular joint in patients with rheumatoid arthritis. Swed Dent J 26:149, 2002 J Oral Maxillofac Surg 63:1371-1374, 2005
Endoscope-Guided Biopsy of a Mandibular Cystic Lesion Michael J. Gliddon, DDS,* Nadarajah Vigneswaran, BDS, DMD,† James J. Xia, MD, PhD,‡ and Jose M. Marchena, DMD, MD§ Proper management of jaw cysts and tumors relies on obtaining an accurate tissue diagnosis. In general, biopsies of cystic lesions jaw are performed by the acquisition of tissue specimens representative of the lining of the lesion. Access to such lesions is generally gained through bony windows in readily accessible
areas of the jaw. Large cystic lesions of the posterior mandible with extension into the ramus, coronoid process, and condyle present special considerations: (1) neoplastic processes may arise from or coexist within the cyst lining, and (2) the acquisition of representative tissue samples from these regions is diffi-
*Former Resident, Department of Oral and Maxillofacial Surgery, The University of Texas Health Science Center, Houston, TX; Major, United States Army Dental Corp, Ft Sill, OK. †Associate Professor, Department of Diagnostic Science, The University of Texas Health Science Center, Houston, TX. ‡Assistant Professor, Department of Oral and Maxillofacial Surgery; Assistant Professor, Division of Pediatric Surgery, Department of Surgery, The University of Texas Health Science Center, Houston, TX. §Assistant Professor, Department of Oral and Maxillofacial Sur-
gery, The University of Texas Health Science Center, Houston, TX; Chief of Oral and Maxillofacial Surgery, Ben Taub General Hospital, Houston, TX. Address correspondence and reprint requests to Dr Marchena: 6516 M.D. Anderson Blvd, Suite DBB 2.059, Houston, TX 77030; e-mail: [email protected] © 2005 American Association of Oral and Maxillofacial Surgeons
0278-2391/05/6309-0019$30.00/0 doi:10.1016/j.joms.2005.05.163
SEVERE ISOLATED TMJ INVOLVEMENT IN JIA
6. Pindborg JJ, Vedtofte P, Reibel J, et al: The calcifying epithelial odontogenic tumor. A review of recent literature and report of a case. APMIS Suppl 1991;23:152 7. Morimoto C, Tsujimoto M, Shimaoka S, et al: Ultrastructural localization of alkaline phosphatase in the calcifying epithelial odontogenic tumor. Oral Surg Oral Med Oral Pathol 56:409, 1983 8. Chomette G, Auriol M, Guilbert F: Tumeur épithéliale odontogéne calcifiée bifocale (Tumeur de Pindborg). Rev Stomatol Chir Maxillofac 85:329, 1984 9. Damm DD, White DK, Drummond JF, et al: Combined epithelial odontogenic tumor: Adenomatoid odontogenic tumor and calcifying epithelial odontogenic tumor. Oral Surg 55:487, 1983 10. Bingham RA, Adrian JC: Combined epithelial odontogenic tumor - Adenomatoid odontogenic tumor and calcifying epithelial odontogenic tumor: report of a case. J Oral Maxillofac Surg 44:574, 1986 11. Takeda Y, Kudo K: Adenomatoid odontogenic tumor associated with calcifying epithelial odontogenic tumor. Int J Oral Maxillofac Surg 15:469, 1986 12. Hicks MJ, Flaitz CM, Wong ME, et al: Clear cell variant of calcifying epithelial odontogenic tumor: Case report and review of the literature. Head Neck 16:272, 1994 13. Melrose RJ: Benign epithelial odontogenic tumors. Semin Diagn Pathol 16:271, 1999 14. Veness MJ, Morgan G, Collins AP, et al: Calcifying epithelial odontogénico (Pindborg) tumor with malignant transformation and metastatic spread. Head Neck 23:692, 2001
15. Cross JJ, Pilkington RJ, Antoun NM, et al: Value of computed tomography and magnetic resonance imaging in the treatment of a calcifying epithelial odontogenic (Pindborg) tumour. Br J Oral Maxillofac Surg 38:154, 2000 16. Slootweb PJ: Bone and cementum as stromal features in Pindborg tumor. J Oral Pathol Med 20:93, 1991 17. El-Labban NG: Cementum-like material in a case of Pindborg tumor. J Oral Pathol Med 19:166, 1990 18. Chomette G, Auriol M, Guilbert F: Histoenzymological and ultrastructural study of a bifocal calcifying epithelial odontogenic tumor. Characteristics of epithelial cells and histogénesis of amyloidlike material. Virchows Arch (Pathol Anat) 403:67, 1984 19. El-Labban NG, Lee KW, Kramer IRH: The duality of the cell population in a calcifying odontogenic tumor (CEOT). Histopathology 8:679, 1984 20. Ai-Ru L, Zhen L, Jian S: Calcifying epithelial odontogenic tumors: A clinicopathologic study of nine cases. J Oral Pathol 11:399, 1982 21. Maiorano E, Altini M, Favia G: Clear cell tumors of the salivary glands, jaws and oral mucosa. Semin Diagn Pathol 14:203, 1997 22. Veness MJ, Morgan G, Collins AP, et al: Calcifying epithelial odontogénico (Pindborg) tumor with malignant transformation and metastatic spread. Head Neck 23:692, 2001 23. Basu MK, Matthews JB, Sear AJ, et al: Calcifying epithelial odontogenic tumor: A case showing features of malignancy. J Oral Pathol 13:310, 1984
J Oral Maxillofac Surg 63:1368-1371, 2005
Severe Isolated Temporomandibular Joint Involvement in Juvenile Idiopathic Arthritis Paolo Scolozzi, MD, DMD,* Geraldine Bosson, MD, DMD,† and Bertrand Jaques, MD, DMD‡ First described by Meyer Diamant-Berger in 1892 and by Frederic Still in 1897, juvenile rheumatoid arthritis represents a heterogeneous group of chronic inflammatory arthritis that begins in childhood and is distinct from adult rheumatoid arthritis.1 In 1997, the International League of Associations for RheumatolReceived from the Department of Otolaryngology/Head and Neck Surgery, Division of Oral and Maxillofacial Surgery, Centre Hospitalier Universitaire Vaudois, Lausanne, Switzerland. *Chief Resident. †Resident. ‡Professor. Address correspondence and reprint requests to Dr Scolozzi: Department of Otolaryngology/Head and Neck Surgery, Division of Oral and Maxillofacial Surgery, Centre Hospitalier Universitaire Vaudois, 1011 Lausanne, Switzerland; e-mail: [email protected] © 2005 American Association of Oral and Maxillofacial Surgeons
0278-2391/05/6309-0018$30.00/0 doi:10.1016/j.joms.2005.05.300
ogy proposed the more universal term of “juvenile idiopathic arthritis” (JIA) and recognized 8 categories: systemic arthritis, oligoarthritis (arthritis affecting 1 to 4 joints in the first 6 months of the disease), extended oligoarthritis, polyarthritis (more than 4 joints in the first 6 months of disease with 2 subgroups, rheumatoid factor (positive or negative), enthesitis related arthritis (largely made up of HLA B-27 related disease), psoriatic arthritis, and “other arthritis.” Juvenile idiopathic arthritis refers to arthritis of unknown cause in 1 or more joints of at least 6 weeks’ duration occurring in children less than 16 years old.2 Juvenile idiopathic arthritis commonly afflicts the temporomandibular joint (TMJ) leading to the destruction of the condylar growth center and subsequent mandibular growth disturbances in severe cases.3-6 This can result in unpleasant facial deformity such as asymmetry, micro- and retrognathia, as well as secondary abnormal dental occlusion development. The ultimate and dramatic consequence of the joint destruc-
1369
SCOLOZZI, BOSSON, AND JAQUES
FIGURE 1. Coronal (A and C) and sagittal oblique (B and D) reconstructions from a thin-slice spiral computed tomography acquisition with bone algorithm through both TMJs shows bilateral extensive flattening of condyles and multiple cortical erosions of the glenoid fossa. Scolozzi, Bosson, and Jaques. Severe Isolated TMJ Involvement in JIA. J Oral Maxillofac Surg 2005.
tion will result in TMJ ankylosis and a definitive limitation of the bite.3-6 We are reporting the case of a 5-year-old girl who presented with isolated bilateral TMJ arthritis without any other articular involvement as exclusive manifestation of a JIA. To the best of our knowledge, only 1 similar case has been reported previously.7
Report of a Case In October 2003, a 5-year-old child was referred by her otolaryngologist for investigation of severe bilateral TMJ pain and trismus of 4 week’s duration. The patient was afebrile and otherwise in good health and extra- and intraoral examinations were normal. The function of the TMJ was evaluated bilaterally for calculation of the Dysfunction Index as described by Hel-
FIGURE 2. Gadolinium-enhanced T1-weighted images revealed a marked thickened inflammatory synovium (pannus) and abundant intra-articular effusion corresponding to synovitis (white arrow) (A-D). Note flattening of the disc otherwise in a normal position (black arrow) (B-D). Scolozzi, Bosson, and Jaques. Severe Isolated TMJ Involvement in JIA. J Oral Maxillofac Surg 2005.
1370 kimo.8 Both TMJs showed signs of severe dysfunction (Di III) with tenderness to palpation of the joint, pain during mouth opening, and limited mobility of the condyles. Mouth opening was restricted to 10 mm and protrusive as well as lateral movements were impossible. There were no spasms in the muscles of mastication and no joint sounds. The otoscopic examination, as well as the rest of the physical examination, was normal. Panoramic radiograph revealed flattening and important bilateral condylar destruction with a narrowed articular space. A complete chemical and microbiological screening revealed no abnormalities. Serological markers were negative for viral hepatitis B and C, Borrelia burgdorferi, Chlamydia psittaci and C. trachomatis, Mycoplasma pneumoniae, and HIV. Rheumatoid factor and antinuclear antibody were negative. The patient underwent a spiral computed tomography acquisition (1 mm slices, pitch 1, overlapping reconstructions every 0.5 mm, bone algorithm) as well as magnetic resonance (MR) examination of the TMJ (head coil, axial 3 mm T2 FSE sequences, axial and coronal 3 mm T1 SE images before and after injection of Gadolinium chelates). Computed tomography and MRI showed bilateral extensive decrease of the joint space as well as a marked flattening of both condyle and glenoid fossa with multiple cortical erosions (Figs 1 and 2). Gadolinium-enhanced T1-weighted images revealed a marked enhancing thickened inflammatory synovium (pannus) and abundant intra-articular effusion. The disc was thinned, but in a normal position at rest and during translation on both sides (Fig 2). In November 2003, given the difficulty in eating caused by the severe limitation of mouth opening as well as the negative response to the NSAID, we decided to perform a bilateral TMJ arthrocentesis with lavage. The procedure was performed under general anesthesia, and 1 mL of slightly hemorrhagic synovial fluid was obtained and the joints were lavaged with 100 mL of NaCl 0.9% using 2 large bore needles. Analysis of all the fluid obtained revealed 200 leucocytes/mm3 and no crystals. Cultures were sterile. The final diagnosis was that of a JIA isolated to the TMJ. The patient’s condition improved rapidly with total disappearance of the TMJ pain within the first 24 hours postoperatively. Physiotherapy with protrusion-opening jaw exercises was started 1 week after the procedure. At the 6-month followup, the patient was pain free and the maximum mouth opening achieved was 40 mm, protrusion movement was 6 mm, right and left lateral movements were 14 mm.
Discussion In the reported case, our patient presented with severe bilateral TMJ arthritis with effusion as well as advanced cortical destruction of both the condyle and the glenoid fossa. In children, 2 main diseases can produce such TMJ lesions: septic arthritis and JIA. Septic arthritis of the TMJ typically affects young children and follows a middle ear infection. Vestigial foramen and fissures between the TMJ and the middle ear, which usually close during the first 2 years of life, account for possible reciprocal spread of inflammation or infection between these 2 organs. Since the introduction of antibiotics, the incidence has been dramatically reduced.9 This diagnosis was ruled out in our patient by the cultures of the synovial liquid, which were sterile.
SEVERE ISOLATED TMJ INVOLVEMENT IN JIA
Although TMJ involvement in children with JIA is not uncommon, it rarely occurs at the beginning of the disease.1-7,10 Other joints such as the knee, hands and feet, hips, and sacroiliac joints are generally involved first. Its prevalence has been reported to be highest in polyarticular, early onset and antinuclear-positive arthritis.4 The most commonly reported symptoms are pain during function and at rest, morning stiffness, restricted maximal voluntary mouth opening, and TMJ crepitation. Patients with JIA of the TMJ can also exhibit functional impairments such as painful TMJ clicking and locking mimicking an internal derangement.5 The prevalence of reported radiologic TMJ abnormalities in children with JIA have varied from 29% to 66% in the literature, with cortical erosions and flattening of the condyle being the most common lesions found.4-6 Svensson et al5 showed that radiographic condylar lesions were significantly correlated with postnormal occlusion, restricted mouth opening, anterior open bite, and mandibular retrognathia. They did not find any significant correlation between condylar lesions and TMJ pain. The main concern in our patient was the isolated involvement of both TMJs without any other articular involvement during 6 months’ follow-up. In such specific cases where the clinical presentation is atypical and unusual, imaging (particularly MRI) is of value in detecting and evaluating intraarticular abnormalities.4-6 MRI is the most sensitive radiologic indicator of disease activity.6 In TMJ involvement, it is more sensitive than clinical evaluation, demonstrating inflammatory changes even in the absence of clinical symptoms.6 In our case, MRI provided the final diagnosis showing the characteristic lesions found in JIA, namely synovial hypertrophy (pannus), intra-articular effusion, and cartilaginous and subchondral bone erosions. Regarding the management of TMJ involvement in JIA, a general consensus concerning treatments does not exist, and they are often extrapolated from those used for other articulations, especially of the knee. Preliminary beneficial effects of simple TMJ arthrocentesis with lavage, without arthroscopy (as proposed by Nitzan et al11) have been reported in adults with rheumatoid arthritis, whereas reports in the pediatric population are lacking. The intra-articular lavage washes the inflamed synovial liquid, which in JIA patients contains different inflammatory proteins and lipid pain mediators.11,12 These proteins and lipids apparently act on nerve endings of the synovial membrane-provoking nociception. The final outcomes of this procedure are decreased pain, increased maximal mouth opening, and improved lateral movements.11,12 Arthroscopy alone or followed by synovectomy has been reported in the orthopedic literature especially for active knee synovitis, whereas only 1 case has
1371
GLIDDON ET AL
been reported for treatment of TMJ involvement in a patient with JIA.7 Intra-articular steroid injections in JIA have been attempted based on the experience with adults.13,14 Although it is a safe and rapidly effective treatment for synovitis, there are no data to confirm whether the disease course is modified in the long term and the literature lacks controlled studies. Its use remains anecdotal. Vallon et al14 found that the long-term development of symptoms and signs from the TMJ and the long-term progression of the joint destruction were low in patients previously treated with steroid intra-articular injections. Despite the good response of our patient after the arthrocentesis and lavage, the management of JIA remains an unsolved dilemma and a great challenge for the future, especially in limiting dramatic growth disturbances.
4.
5.
6. 7.
8.
9.
10. 11.
12.
References 1. Schneider R, Passo MH: Juvenile rheumatoid arthritis. Rheum Dis Clin North Am 28:503, 2002 2. Petty RE, Southwood TR, Baum J, et al: Revision of the proposed classification criteria for juvenile idiopathic arthritis: Durban, 1997. J Rheumatol 25:1991, 1998 3. Svensson B, Larsson A, Adell R: The mandibular condyle in juvenile chronic arthritis patients with mandibular hypoplasia:
13.
14.
A clinical and histological study. Int J Oral Maxillofac Surg 30:300, 2001 Pedersen TK, Jensen JJ, Melsen B, et al: Resorption of the temporomandibular condylar bone according to subtypes of juvenile chronic arthritis. J Rheumatol 28:2109, 2001 Svensson B, Adell R, Kopp S: Temporomandibular disorders in juvenile chronic arthritis patients. A clinical study. Swed Dent J 24:83, 2000 Johnson K, Gardner-Medwin J: Childhood arthritis: Classification and radiology. Clin Radiol 57:47, 2002 Martini G, Bacciliero U, Tregnaghi A, et al: Isolated temporomandibular synovitis as unique presentation of juvenile idiopathic arthritis. J Rheumatol 28:1689, 2001 Helkimo M: Studies on function and dysfunction of the masticatory system. II. Index for anamnestic and clinical dysfunction and occlusal state. Swed Dent J 67:101, 1974 Scolozzi P, Becker M, Richter M: Temporomandibular joint osteoarthritis: A cause of a serous otitis media? A case report. J Oral Maxillofac Surg 62:97, 2004 Kulas DT, Schanberg L: Juvenile idiopathic arthritis. Curr Opin Rheumatol 13:392, 2001 Nitzan DW, Dolwick MF, Martinez GA: Temporomandibular joint arthrocentesis: A simplified treatment for severe, limited mouth opening. J Oral Maxillofac Surg 49:1163, 1991 Trieger N, Hoffman CH, Rodriguez E: The effect of arthrocentesis of the temporomandibular joint in patients with rheumatoid arthritis. J Oral Maxillofac Surg 57:537, 1999 Cleary AG, Murphy HD, Davidson JE: Intra-articular corticosteroid injections in juvenile idiopathic arthritis. Arch Dis Child 88:192, 2003 Vallon D, Akerman S, Nilner M, et al: Long-term follow-up of intra-articular injections into the temporomandibular joint in patients with rheumatoid arthritis. Swed Dent J 26:149, 2002 J Oral Maxillofac Surg 63:1371-1374, 2005
Endoscope-Guided Biopsy of a Mandibular Cystic Lesion Michael J. Gliddon, DDS,* Nadarajah Vigneswaran, BDS, DMD,† James J. Xia, MD, PhD,‡ and Jose M. Marchena, DMD, MD§ Proper management of jaw cysts and tumors relies on obtaining an accurate tissue diagnosis. In general, biopsies of cystic lesions jaw are performed by the acquisition of tissue specimens representative of the lining of the lesion. Access to such lesions is generally gained through bony windows in readily accessible
areas of the jaw. Large cystic lesions of the posterior mandible with extension into the ramus, coronoid process, and condyle present special considerations: (1) neoplastic processes may arise from or coexist within the cyst lining, and (2) the acquisition of representative tissue samples from these regions is diffi-
*Former Resident, Department of Oral and Maxillofacial Surgery, The University of Texas Health Science Center, Houston, TX; Major, United States Army Dental Corp, Ft Sill, OK. †Associate Professor, Department of Diagnostic Science, The University of Texas Health Science Center, Houston, TX. ‡Assistant Professor, Department of Oral and Maxillofacial Surgery; Assistant Professor, Division of Pediatric Surgery, Department of Surgery, The University of Texas Health Science Center, Houston, TX. §Assistant Professor, Department of Oral and Maxillofacial Sur-
gery, The University of Texas Health Science Center, Houston, TX; Chief of Oral and Maxillofacial Surgery, Ben Taub General Hospital, Houston, TX. Address correspondence and reprint requests to Dr Marchena: 6516 M.D. Anderson Blvd, Suite DBB 2.059, Houston, TX 77030; e-mail: [email protected] © 2005 American Association of Oral and Maxillofacial Surgeons
0278-2391/05/6309-0019$30.00/0 doi:10.1016/j.joms.2005.05.163