Chondrosarcoma of the temporomandibular joint: a case report and review of the literature

Chondrosarcoma of the temporomandibular joint: a case report and review of the literature

454 Abstracts oral melphalan and prednisone initially and thalidomide afterwards was noneffective and toxic. Instead, plasmapharesis resulted in decr...

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454 Abstracts

oral melphalan and prednisone initially and thalidomide afterwards was noneffective and toxic. Instead, plasmapharesis resulted in decrease of the total levels and the monoclonal portion of the IgG paraprotein. At 6-month follow-up, no progression of the oral nodules or development of new amyloid deposits elsewhere was noted. Conclusions. This case exemplifies a rare clinical presentation of progression of MGUS to primary amyloidosis with lesions first discovered and, at the time of the last follow-up, effectively limited to the oral cavity.

SELF-ASSESSMENT OF TRAINING TO PERFORM ORAL CANCER PREVENTION AND DETECTION: A COMPARISON OF PROVIDER GROUPS. L. L. Patton, T. E. Ashe, J. R. Elter, J. H. Southerland, R. P. Strauss, University of North Carolina, Chapel Hill, NC. Background. North Carolina (NC) has increasing incidence and stable high mortality rates of oral and pharyngeal cancer (OPC) over the 10 years 1990 to 1999, as compared to other states. NC ranks fourth worst in dentist-to-population ratio among the 50 states and DC (December 31, 1999) and 22nd worst in primary care physician–topopulation ratio (December 31, 2000). Objective. The purpose of this project is to assess and compare the self-reported adequacy of training in OPC early intervention activities among 4 healthcare provider groups. Study design. Between May 2002 and May 2003, 4 pretested surveys were mailed to random samples of the states’ licensed dentists (n = 1115), dental hygienists (n = 1223), family physicians (n = 1058), and nurse practitioners (n = 967) practicing in NC. Effective response rates after 2-3 mailings ranged from 53% to 26% depending on provider group, with higher response rates from dental providers. Bivariate associations were explored for association between provider type and agreement on questions pertaining to adequacy of professional training to provide tobacco and alcohol cessation education and to examine patients for oral cancer and palpate neck nodes. Results. Of 1235 dental providers (584 dentists and 651 hygienists) and 567 medical providers (273 family physicians and 294 nurse practitioners), there was high agreement (93% and 92% respectively) with the statement that early detection improves 5-year survival rates from oral cancer, with no significant difference between groups (P = .39). Compared to medical providers, dental providers were significantly less likely to feel adequately trained to conduct tobacco cessation (33%) and alcohol cessation (15%) education and to palpate neck nodes (70%; odds ratios [ORs] = 0.11, 0.09, 0.06, respectively; all P\.0001) but were significantly more likely to feel adequately trained to perform oral cancer examinations (81%; OR = 4.47; P\.0001). Among dental providers, those who felt they had adequate training in tobacco cessation education were significantly (P\.05) more likely to assess past (OR = 1.90), present (OR = 2.04), and type and amount (OR = 1.51) of tobacco on patient medical histories. Similarly, dental providers who felt adequately trained in alcohol cessation had over twice the likelihood of assessing alcohol (OR = 2.33 for current use; OR = 2.99 for past use, and OR = 2.43 for type and amount) on medical histories than did providers who felt inadequately trained. Conclusions. Dental providers in NC more frequently recognize deficits in their training regarding tobacco and alcohol cessation education for patients than do medical colleagues; however, they feel better trained to effectively examine patients for oral cancer. Training program curricular enhancements and continuing education courses are needed to prepare dental providers in NC to undertake these important prevention and health promotion activities that may impact increasing OPC incidence rates. Supported by NIH R21 DE14413.

ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY April 2004 ¨ GREN’S SYNDROME: A PILOTETANERCEPT IN SJO RANDOMIZED, DOUBLE-BLIND, PLACEBO-CONTROLLED CLINICAL TRIAL. V. Sankar, M. T. Brennan, M. R. Kok, R. A. Leakan, J. A. Smith, J. Manny, B. J. Baum, S. R. Pillemer, National Institutes of Health, Bethesda, Md, Carolinas Medical Center, Charlotte, NC, and University of Amsterdam, The Netherlands. Objective. To assess the safety and potential efficacy of etanercept in the treatment of Sjo¨gren’s syndrome (SS). Study design. This pilot study was a 12-week, randomized, placebo-controlled, double-blind trial of etanercept with 14 subjects in each group. Patients received 25 mg of etanercept or placebo by twice weekly subcutaneous injections. Patients met American-European classification criteria for SS. The primary outcome required at least 20% improvement from baseline values for at least 2 of 3 of the following: 1) subjective or objective measures of dry mouth; 2) subjective or objective measures of dry eyes; 3) IgG or ESR. Results. Of the 14 subjects on etanercept, 11 had primary SS and 3 had SS secondary to RA. Mean baseline measures did not differ between groups. Three etanercept and 1 placebo patient did not complete the trial. Five etanercept and 3 placebo subjects showed improvement at 12 weeks compared with baseline in the primary outcome, which was not significant (P = .2). No significant differences occurred between the groups for changes over the trial in subjective oral or ocular measures (visual analog scales), IgG, Schirmer I test, Van Bijsterveld scores, or salivary flow. ESR decreased in the etanercept group after adjusting for baseline levels (P = .0147). However, the level of improvement was not clinically meaningful (18.6%). Conclusions. 1) Treatment with etanercept 25 mg twice weekly for 12-weeks was not beneficial in SS. 2) No clinically meaningful improvement was noted for any outcomes.

CHONDROSARCOMA OF THE TEMPOROMANDIBULAR JOINT: A CASE REPORT AND REVIEW OF THE LITERATURE. E. Sarlani, N. G. Nikitakis, R. Fernandes, R. A. Ord, University of Maryland, Baltimore. Objective. Chondrosarcoma of the temporomandibular joint (TMJ) is very rare with less than 20 cases reported in the literature. The purpose of this study is to present a case of chondrosacroma of the left TMJ and to review the previously reported cases in order to elucidate the clinical and radiographic features, the histopathologic characteristics, and the treatment and prognosis of this neoplasm. Case study. Our patient, a 42-year-old Caucasian male, presented with a complaint of swelling in the left preauricular area. In addition, the patient had noticed tingling of his left cheek and a limitation of his mouth opening. Clinical examination revealed a firm swelling approximately 2 3 3 cm just anterior to the left tragus. Computed tomography (CT) demonstrated a well rounded mixed radiolucent-radiopaque lesion of the left mandibular condyle with surrounding soft tissue changes. Abnormal increased uptake in the left TMJ area was seen on a bone scan. A biopsy of the left condylar region was performed, rendering a diagnosis of benign osteochondroma, which was treated with resection of the left condyle. Six months later the patient presented with recurrent left condylar swelling. Clinical examination revealed a 3 3 4 cm firm swelling over the left TMJ, extending behind the ear and elevating the ear lobe. CT showed marked periosteal ossification and dystrophic soft tissue calcification around the left mandibular condyle. A biopsy was done, and histopathologic examination revealed malignant features diagnostic of chondrosarcoma, grade II. The patient underwent left total parotidectomy and left hemimandibulectomy from the first molar to the base of the skull, which was followed by high-dose radiation therapy. Treatment resulted in a significant facial deformity

ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY Volume 97, Number 4 including a complete paralysis of the left facial nerve. In the course of the following years the patient underwent several reconstruction procedures. No signs of recurrence were noted at the 7-year follow-up. Conclusions. Chondrosarcoma of the TMJ typically presents with preauricular swelling, while pain and trismus may also be present, mimicking common benign TMJ disorders. Differentiation from these conditions is of paramount importance, because an early and accurate diagnosis of chondrosarcoma followed by prompt and appropriate treatment will significantly improve the prognosis.

THE CLAYTON INTRAAURAL DEVICE FOR TEMPOROMANDIBULAR DISORDERS. T. P. Sollecito, L. G. Clayton, S. S. DeRossi, L. Laster, M. S. Greenberg, University of Pennsylvania, Philadelphia. Objective. Temporomandibular disorders (TMDs) are a group of conditions characterized by chronic pain and dysfunction in the temporomandibular joint (TMJ) and the muscles of mastication. The current clinically effective, reversible treatments for TMDs include physical therapy, behavioral therapy, pharmacological therapy, and intraoral splint therapy. The Clayton Intraaural Device (CID) is a hollow plastic patented device which rests in the outer third of the ear canal when the jaw is slightly opened. This device may add to the clinically effective, safe and reversible, conservative treatments for TMDs. The following pilot study was designed to test safety and efficacy of the CID and power a larger study. Study design. A single group study was used to determine the differential response to using the CID across a 16-week period, including 4 weeks of pretreatment observation. Subjects were recruited from those patients experiencing jaw pain/dysfunction and who presented to the Department of Oral Medicine at the University of Pennsylvania. Subjects who had a history of direct jaw trauma, a history of connective tissue disease, or received treatment for TMD with an occlusal splint in the past 6 months were excluded. A history of jaw pain and/or dysfunction was determined and its severity rated using a patient-reported visual analog scale (VAS) ranging from 0 (no pain) to 10 (most severe pain). Each subject was considered having a TMD if they had the presence of 1 or more of the following findings associated with pain (VAS > 4): increased ($60 mm) or decreased (#40 mm) range of interincisal jaw opening (VAS > 4), pain upon any jaw movement (VAS > 4), pain on digital palpation (1 lb. of pressure) of the periauricular area or external auditory meatal areas (VAS $ 4), pain on digital palpation (1 lb. of pressure) in 2 or more muscles of mastication (VAS $ 4), and joint sound with pain (VAS > 4). Subjects with absence of all the listed findings were considered normal. Subjects diagnosed with a TMD were invited to enroll in the study. Subjects who enrolled then completed a 4-week observation period, during which the subject completed VAS 2 times daily; subjects with a mean VAS > 4 were permitted to enter in the in the treatment phase of the study. An ear impression was then taken with the jaw slightly opened. During the treatment phase of the study, subjects were assessed and reassessed for TMD using 4 indices: VAS, Craniomandibular Index (CMI), Symptom Severity Index (SSI) and the North Carolina TMJ Scale. Results. Of the initial 15 subjects enrolled in the treatment phase of the study, 9 subjects wore the CID. Of these subjects, one completed 2 months of follow-up and 8 completed the full 3-month study period. The VAS values collected monthly from the home VAS diaries revealed that the study subjects had a 55% average reduction (P\.004) in TMD pain from pretreatment to 3 months posttreatment. The CMI data showed that the study subjects had a 65% average reduction (P\.001) in TMD pain and dysfunction from pretreatment to 3 months posttreatment. The subjective assessment of subject pain, the SSI, showed a 42% reduction (P\.003), in pain from pretreatment to 3 months post treatment. Of the 8 TMJ Scales reports analyzed from pre-

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to posttreatment, all revealed global (TMJ) score reduction, suggesting some resolution of the TM symptoms. Additionally the self-reported TMJ Scales’ ‘‘pain report’’ and ‘‘palpation pain’’ scores also decreased from the start of treatment to the end of treatment in all cases. This validated assessment tool suggests clinical improvement in all 8 cases. All of the 8 subjects who completed the study had their hearing tested with and without the CID. There was no significant change in hearing with the use of the CID. Conclusions. In our pilot study, we demonstrated that treatment with the CID leads to an overall reduction of the pain and dysfunction of temporomandibular disorders. From this pilot data, the CID may be a safe, effective modality for the treatment of TMD. A larger randomized controlled clinical trial is being planned.

ASSOCIATION BETWEEN GENE POLYMORPHISMS AND PAIN SEVERITY IN PATIENTS WITH RECURRENT APHTHOUS STOMATITIS (RAS). M. H. Thornhill, L. Baccaglini, M. Bazrafshani, W. Ollier, University of Texas Health Science Center, San Antonio, and University of Manchester, UK. Objective. Recurrent aphthous stomatitis (RAS) is a painful oral inflammatory condition characterized by recurrent episodes of oral mucosal ulcerations. Because there is considerable variation in the level of pain experienced by different patients with RAS, we determined whether certain genetic factors (i.e., inflammatory pain–related cytokine gene polymorphisms) are associated with the intensity of orofacial pain. Study design. We enrolled 100 Caucasian patients with idiopathic minor RAS and 91 controls. Data were collected through blood samples (for genotyping), clinical examinations (to confirm the RAS diagnosis), and patient diaries (for data on pain level, ulcer size, and number). A composite pain index (PI) adjusted for ulcer size and number was calculated for each individual and overall. Patients were assigned to high and low pain groups based on the mean PI score cutoff. DNA was extracted from blood samples and purified. Genotyping was performed to identify the following gene polymorphisms: IL-1B-511, IL6-174, IL-1A-889, IL-1B +3954, and TNFA-308. After confirming HardyWeinberg equilibrium, Mantel-Haenszel chi-square or Fisher’s exact test and logistic regression (SAS version 8.2) were used to calculate P-values and odds ratios (OR) with 95% confidence intervals (95% CI) for the association between the genotype or allele and orofacial pain level (none, low or high). Results. RAS patients in the high pain group had 4.35 times the odds (95% CI = 2.55-7.41) of having inherited the G allele of IL-1B511 compared to controls. RAS patients in the low pain group had 1.55 times the odds (95% CI = 0.94-2.56) of having inherited this allele compared to controls. RAS patients in the low (OR = 2.06; 95% CI = 1.23-3.51) and high (OR = 3.65; 95% CI = 1.92-6.93) pain groups were also more likely to have inherited the G allele of IL-6-174 compared to controls. When directly comparing high vs. low RAS pain, the IL-1B-511 and the IL-6-174 G/G genotype was associated with higher RAS pain (Table I).

Table I. Genotype associations between RAS patients with high/low pain Pain level Polymorphism Genotype Low High IL-1B-511 IL-6-174

A/G, A/A G/G G/C, C/C G/G

45 9 23 31

28 18 13 33

OR (95% CI)

x2 (P-value)

Ref 6.30 (0.012) 3.21 (1.27-8.14) Ref 2.19 (0.139) 1.88 (0.81-4.36)