Trismus and preauricular swelling in a 20-year-old black woman

J Oral Maxillofac Surg 54:1234-1239, 1996

Trismus and Preauricular Swelling 20-Year-Old Black Woman SALVATORE

L. RUGGIERO, DMD, MD,* EILEEN HILTON, AND THOMAS W. BRAUN, DMD, PhD*

MD,*

mouth, and palate were normal with no evidence of a deviated uvula. The dcntition was grossly intact. The initial laboratory values were significant for a white ccl1 count of 9,700 cells/mm3 with a normal differential count, an hematocrit of 37.9%, and a platelet count of 397,000 cells/mm’. The panoramic radiograph did not show any carious teeth, but the left mandibular condyle region appeared radiolucent compared with the contralateral side (Fig 1). An axial and coronal computed tomography (CT) scan were obtained that showed a large subperiosteal fluid collection along the medial and lateral aspects of the mandibular ramus extending from the angle of the mandible to the condyle (Fig 2). Superiorly this process clearly involved the joint cavity and was associated with destruction of the superior and lateral aspect of the mandibular condylc (Fig 3). The paranasal sinuses, mastoids processes, and parotid glands were normal.

Case Presentation A 22-year-old Haitian woman was referred to our institution with a 6-week history of trismus and preauricular swelling. The patient had initially presented to a dentist after experiencing pain and “stiffness” of her jaw on the left side. A diagnosis of myofascial pain syndrome was made, and the patient was treated with a soft diet and a nonsteroidal antiinflammatory agent. However, over the course of the next 3 weeks the pain and trismus progressively worsened and was now associated with a left preauricular swelling. The patient was referred to an otolaryngologist, who made the presumptive diagnosis of acute parotitis and placed the patient on a first generation cephalosporin. During the following 2 weeks, the left facial swelling and pain worsened significantly despite her compliance wilh the antibiotic regimen. At that time, she presented to our institution for further evaluation. The patient dcnicd any history of fever, chills, odynophagia, or recent trauma. Her medical history was unremarkable. She was born in Haiti and emigrated to the United States at the age of 6. Since then she has not traveled outside the United States. She denied owning any pets and did not abuse drugs. She was employed as a ward clerk in a New York City hospital The patient presented with normal vital signs and an oral temperature of 99.8”F. The clinical examination was significant for a large, tender, erythematous swelling of the left preauricular region extending from the zygomatic arch to the angle of the left mandible. Mandibular opening was limited to 10 mm and was associated with crepitus in the left temporomandibular joint. Thcrc was no cervical adcnopathy detected on palpation. Clear saliva flowed easily from the left Stenson’s duct. Results of an otoscopic examination of the left ear were normal. The oral mucosa, tongue, floor of

Differential Thomas W. Braun,

DMD,

Diagnosis PhD

The case described presents the clinician with several avenues for consideration. In light of the history and clinical and radiographic findings, the differential diagnosis must include neoplastic and infectious processes. Benign neoplastic processes of the temporomandibular joint that must be considered include osteochondroma, synovial chondromatosis, pigmented villonodular synovitis, chondroblastoma, osteoblastoma, chondroma, and ganglion cyst. Malignant processes include osteosarcoma and chondrosarcoma. The radiographic findings did not include the presence of calcified particles (joint mice) within the joint space. However, even though synovial chondromatosis is characterized by joint mice, it cannot be discounted by their radiographic absence. Radiolucency of the condyle, with erosion, would cause concern for malignant processes such as osteosarcoma and chondrosarcoma. The patient falls in an age range in which this is more likely. The far more rare pigmented villonodular synovitis, although a consideration, would be low on the list of differential diagnoses. The excessive elaboration of fluid would lead one to consider the possibility of a ganglion cyst. However, the radiographic

Received from the Long Island Jewish Medical Center, New Hyde Park, NY. * Assistant Attending, Department of Dental Medicine, Division of Oral and Maxillofacial Surgery. -1Attending, Department 01 Medicine, Division of Infectious Deseases. $ Professorand Chairman of Oral and Maxillofacial Surgery, University of Pittsburgh, Pittsburgh, PA. Address correspondenceand reprint requeststo Dr Ruggiero: Department of Dental Medicine, Division of Oral and Maxillofacial Surgery, Long Island Jewish Medical Center, New Hyde Park, NY 11040. 0 1996 American Association of Oral and Maxillofacial 0278.2391/96/5410-0012$3.00/O

in a

Surgeons

I234

RUGGIERO,

HILTON,

1235

BRAUN

FIGURE 1. Panoramic radiograph demonstrating an intact tion and subtle radiolucent areas in the left condyle.

denti-

characteristics of chondroblastoma, osteoblastoma, or chondroma were not present. Infectious processes to be considered include acute septic arthritis, osteomyelitis, and conceivably chronic granulomatous diseases. The persistence of a swelling that is unresponsive to antibiotics, with no evidence of fluctuance and accompanied by a relatively lowgrade fever and normal laboratory findings, would tend to preclude an acute infectious process. Nonetheless, with a deeply centered infection, penetration by antibiotics may require extended parenteral delivery. A history of direct trauma or injection into the joint or acute infection of the external auditory canal adjacent to the joint, does not exist and would tend to diminish the likelihood of such conditions. Osteomyelitis is possible, but with no active source of infection or specific history becomes less likely. Although the laboratory

FIGURE 3. Coronal CT scan of the left mandibular ramus temporomandibular joint that shows destruction of the superior lateral aspects of the mandibular condyle.

and and

studies did not include a tuberculin skin test, it should be considered based on the patient’s geographic background. Tuberculosis occurring alone or as a sequela of human immunodeficiency virus (HIV) infection must be suspected. Because foreign-born individuals have been shown to be more likely to have tuberculosis, the country of origin should be considered. Haiti is one of several countries that has a significantly high rate of tuberculosis. Nonetheless, the occurrence of tuberculous arthritis of the temporomandibular joint is extremely rare and, although it has to be considered, may not be first in the differential diagnosis. In summary, the presence of a destructive process of the mandibular condyle associated with prominant swelling unresponsive to antibiotics and contributing to trismus and limited mandibular function in the absence of otologic or salivary gland pathology would lead one to consider a differential diagnosis of neoplastic and infectious processes. Those neoplastic processes are pigmented villonodular synovitis, synovial chondromatosis, ganglion cyst, chondroblastoma, osteoblastoma, chondroma, osteosarcoma, and chondrosarcoma. Those infectious processes to be considered are acute suppurative arthritis, osteomyelitis, and chronic tuberculous arthritis. Exploration and biopsy are necessary to establish the definitive diagnosis. SUBSEQUENT COURSE

FIGURE 2. Axial CT scan of the left mandibular ramus is significant for a large subperiosteal fluid collection along the medial and lateral aspects of the left mandibular ramus that extends from the angle of the mandible to the condyle.

The patient was brought to the operating room, where an intraoral incision and drainage was performed. An incision was made along the left ascending ramus, and the periosteum

1236 was reflected from the lateral and medial aspect of the mandibular ramus. Approximately 10 mL purulent drainage was obtained. A ‘/.-inch Penrose drain was placed on the medial and lateral aspects of the ramus. Specimens for a Gram stain, routine culture, and culture for acid-fast bacteria, fungus, and mycobacteria were submitted to microbiology. The patient was placed on intravenous penicillin and metronidazole. She did well postoperatively and was discharged on the second postoperative day on oral penicillin and metronidazole. The patient returned 1 week later with recurrent preauricular swelling, pain, a fever of 100.7”F, and a white blood cell count of 10,700 cells/mm3. The chest radiograph was clear. A repeated CT scan of the head and neck was performed that showed a fluid collection lateral to the temporomandibular joint associated with lytic changes in the condylar head and neck. The patient was admitted and this area was explored transorally. An incision and drainage was performed in addition to a curettage of the left lateral ramus and condylar neck region. The specimens obtained were submitted to pathology and microbiology. Culture results from the initial drainage procedure showed mixed oropharyngeal flora, including coagulase-negative staphylococcal and Actinomyces israeli species. The acidfast bacterial smear was negative and all fungal and mycobacterial cultures were still pending. Based on these results, and the poor clinical response to penicillin and metronidazole, the patient was placed on ampicillin/sulbactam. This antibiotic regimen was changed to intravenous doxycycline because of an adverse reaction to the ampicillin/sulbactam preparation. Despite a decrease in the white blood cell count and fever, the preauricular region remained swollen and tender. Additional laboratory tests showed a negative antinuclear antibody level, negative rheumatoid factor, and a nonreactive rapid plasma reagin (RPR) test. The patient’s HIV status was negative. The microscopic evaluation of the specimen submitted showed necrotic bone with no evidence of granulomas or an underlying neoplasm. A purified protein derivative (PPD) test was performed on the fourth postoperative day, which was interpreted as markedly positive. Despite the lack of definitive culture data, the patient was placed on antituberculous chemotherapy on the fifth postoperative day. Shortly thereafter, the swelling and tenderness began to resolve, the mandibular range of motion increased, and the erythrocyte sedimentation rate (ESR) began to normalize. On the 15th postoperative day, the patient was discharged on intravenous doxycycline, oral rifampin, PZA, INH, and ethambutol. Approximately 4 weeks after the initial incision and drainage, the culture data confirmed the growth of M tuberculosis. The antibiotic sensitivity tests, which were done at the National Jewish Center for Immunology and Respiratory Medicine in Denver, Colorado, showed M tuberculosis test was 100% resistant to INH, ethionamide, PZA, streptomycin, and kanamycin. Additional sensitivity testing showed that the organism was moderately susceptible to ofloxacin, ethambutol, capreomycin, cycloserine, and para-aminosalicylic acid (PAS). Accordingly, the antibiotic regimen was changed to olfloxacin, ethambutol, ,capreomycin, and PAS. The patient is now 15 months from the initiation of treatment and is symptom free, with no evidence of disease. The ESR, which was markedly elevated at 120 mm/hr at the onset of treatment, had returned to 24 mm/hr. A chest radiograph performed 9 months after the diagnosis was established was still normal. The patient’s interincisal opening was 40 mm, with a minimal amouniof deviation and no associated pain. A CT scan and panoramic radiograph of the left temporomandibular joint and posterior mandible showed remodeling

TRIMUS

AND

SWELLING

IN A 20.YEAR-OLD

of the condyle with a normal-appearing oid fossa (Figs 4 and 5).

WOMAN

joint space and glen-

Discussion Most, if not all, infections of the maxillofacial region are polymicrobial in nature, especially those of dentoalveolar origin. This fact is not surprising in light of the enormous number and variety of microorganisms that normally inhabit the oropharynx and paranasal sinuses. It has been estimated that 10” bacteria per gram of tissue normally reside within the extended environment of the oral cavity, representing more than 265 bacterial species or groups.‘-4 There is no doubt that our understanding of this complex microbiota was greatly enhancedwith the advent of more sophisticated collection and culturing techniques for anaerobic bacteria. Despite the large numbers of bacteria within the oral cavity, these organisms are not pathogenic, provided that the mucosal barrier is intact and the host is immunocompetent. These “normal” flora are in a way protective to the host in that they create a microenvironment

that

allows

certain

bacteria

to flourish

while

restricting the growth of others. Factors that disrupt this delicate balance can result in the development of a clinical infection. The patient in this casepresented with a history and physical findings that were not typical for a routine dentoalveolar infection. In fact, it is likely that this infectious processhad been establishedmonths before her presentation to our facility. Despite the physical evidence of abscessformation, there was no obvious dentoalveolar sourceby clinical or radiographic examination. The most revealing feature was the amount of bony destruction of the mandibular condyle that was noted on the admission CT scan. Nevertheless, the presence of a large abscesscavity is always somewhat comforting to the clinician, because surgical drainage will most often expedite recovery. The lack of significant improvement

FIGURE showed

in this

patient

after

4. Panoramic radiograph 9 months some degree of condylar remodeling.

an adequate

into treatment

that

RUGGIERO,

FIGURE treatment modeling

HILTON,

BRAUN

5. Coronal CT scan of the left condyle 9 months into clearly demonstrates a significant amount of condylar reand a normal joint space and glenoid fossa.

drainage procedure, coupled with the absence of organisms on the original Gram stain and culture, prompted an extended investigation for an atypical organism or an immunocompromised host. At this point in the treatment algorithm, the antibiotic regimen must be significantly broadened until such time as the patient significantly improves or the causative organism is identified. A PPD test, which was done on her second hospital admission, was grossly positive. Despite the fact that no acid-fast bacteria were isolated on the Gram stain: this was considered sufficient evidence to initiate treatment for M tuberculosis. It is very unusual for M tuberculosis to present as a co-pathogen in a polymicrobial mandibular infection within an immunocompetent host. Although there have been many reports of polymicrobial infections involving M tuberculosis, nearly all cases represent pulmonary infections in an immunocompromised patients .5-7The patient in this case was HIV negative and had no clinical or laboratory evidence of a compromised immune system. There are few reported cases of tuberculous osteomyelitis of the mandible and associated soft tissues,8-2o with one study citing an incidence of mandibular involvement of less than 2% of all musculoskeletal tuberculosiszl In nearly all of these cases, the patient either had a concurrent tuberculous infection elsewhere (ie, lungs and long bones) or the patient had a history of previous infection. The origin of such infections remains debatable and must take into consideration the mode of exposure (aerosolized organisms, infected sputum, hematogenous spread) and the mode of inocu-

1237 lation (mucosal breech). In most of the reported cases of oral tuberculosis infection the vector of exposure was through expectorated, infected sputum, with the portal of entry being a periodontal defect, a carious tooth, a recent extraction socket, or a mandibular fracture site. The incidence of primary extrapulmonary tuberculosis has been well described.22,23 It has been reported to represent 17% of all reported cases of M tuberculosis infections in Canada” and as much as 30% of such infections in the United Statesz3 Of those cases representing extrapulmonary tuberculosis, 6% to 15% were primary infection of the bone and joints, none of which involved the temporomandibular joint.22*23 In contradistinction, the occurrence of primary intraoral/mandibular tuberculous infection, in which the oral cavity is proved to be the only site of involvement, is extremely uncommon.24-26 In retrospect, it is clear that the patient had involvement of the temporomandibular joint early in the course of her clinical infection, given the initial symptoms of preauricular pain and the extent of condylar destruction on the radiograph. This would imply that the infection may have been initiated in the condyle or joint space and infected the surrounding hard and soft tissues secondarily. There are no reported cases of primary tuberculous arthritis of the temporomandibular joint in the English literature. The cause of this particular infection remains unclear. One could speculate that a subclinical M tuberculosis infection of a hematologic or lymphatic nature existed, which subsequently inoculated the hard tissues of the right mandible. In fact, most forms of nonpulmonary primary tuberculosis (including tuberculous osteomyelitis) are thought to emanate from hematogenous dissemination soon after the primary infection occurs.27 The common involvement of weight-bearing joints and vertebral bodies with such infections may be related to the propensity of M tuberculosis to occur in areas of previous trauma. However, such a scenario does not explain why other much more likely sites of seeding (ie, lung and kidney) were not involved initially or subsequently. Another possible mode of oral infection is by direct inoculation though infected food or by a contaminated object (ie, eating utensil). This would require that a violation of the mucosal barrier be present. In the patient in this report, there were no such violations in the integument to account for a vector of infection. Other than the type of antimicrobial agent, the current treatment of tuberculous osteomyelitis is not unlike that for osteomyelitis caused by other, more typical, organisms (eg, Actinomyces and Staphylococcus). It involves a long course of antibiotics, with surgical excision or decortication reserved for the most refractory cases. In the preantibiotic era, surgical intervention (ie, amputation and arthrodesis) was more com-

1238 mon because of the relative paucity of other effective therapeutic modalities. Both tuberculous and nontuberculous osteomyelitis should be approached with equal diagnostic fervor. An adequate biopsy and culture is absolutely required to provide the histologic and microbiologic evidence needed to make an accurate diagnosis.““’ This approach is particularly relevant, considering the fact that culture results for mycobacterium can take as long as 4 weeks to be completed. Histologic evidence of granulomatous changes in bone or joint tissue in such patients provides an added insight as to the atypical infection, prompting the clinician to consider a mycobacterial process and begin appropriate chemotherapy in a more timely fashion. In general, once the diagnosis of tuberculous osteomyelitis has been made, the initiation of antituberculous chemotherapy (the selection of which is based on culture and antibiotic sensitivity profiles) is very effective in most cases. The chemotherapeutic regimen for extrapulmonary tuberculosis is the same as that for pulmonary tuberculosis. The number and type of drugs selected, as well as the duration of therapy, depend on several variables, including the gross number of bacilli found in the lesion, the compliance of the patient, the presence of drug-resistant organisms, the presence of a life-threatening infection, and the immune status of the patient3’ Provided that there are no resistant organisms, initial therapy usually consists of rifampin and isoniazid for either a 6- or a 9-month period. Other agents, such as streptomycin, ethambutol, pyrazinamide, and pyridoxine, can be added in those cases involving recurrent disease or multidrug-resistant organisms.32 In general, surgical treatment for this disease (ie, pulmonary resection, bone debridement, arthrodesis) is of limited efficacy and only indicated in those instances in which intense chemotherapeutic regimens have failed. The role of M tuberculosis and other opportunistic organisms should not be overlooked in complicated head and neck infections given the ever-increasing population of immunocompromised patients. Recently there has been a resurgence of tuberculosis within the inner-city population that is becoming a growing public health concern.33 In New York City alone, the incidence of reported cases of tuberculosis has increased 132% from 1980 to 1990.33 This is coupled with an increasing incidence of extrapulmonary tuberculous infections.34 These figures seem to be highest in the inner-city and urban environments, where the overall number of immigrants continues to grow. From an epidemiologic standpoint, these statistics become more significant when one considers recent data that have shown tuberculosis incidence rates in the immigrant population to be four times that of native-born United States citizens.35X36

TRIMUS

AND

SWELLING

IN A 20-YEAR-OLD

WOMAN

Of particular concern is the emergence of a multidrug-resistant strain of tuberculosis in these same large urban areas. In the United States this represents an increase in prevalence from 2% to 9% over the past three decades.37 In those patients with multi-drug-resistant organisms, the most powerful predictors of such an infection are a history of previous treatment for tuberculosis and HIV infection. Although the patient presented in this report did not have a history of tuberculosis or HIV infection, her risk and potential for exposure was high (ie, employment at a New York City hospital and emigration from Latin America). References 1. Chow AW, Roser SM, Brady FA: Orofacial odontogenic infections. Ann Intern Med 88:392, 1978 2. Gordon DF, Stutman M, Loesche WJ: Improved isolation of anaerobic bacteria from the gingival crevice area of man. Appl Microbial 21: 1046, 197 1 3. Moore WEC, Ranney RR, Holdman LV: Subgingival miroflora in periodontal disease: Cultural studies, in Genco RJ, Mergenhagen S (eds): Host-parasite Interaction in periodontal Disease. Washington, DC, American Society for Microbiology. 1982, pp 13-26 4. Gill Y, Scully C: Orofacial odontogenic infections: Review of microbiology and current treatment. Oral Surg Oral Med Oral Path01 70:155, 1990 5. Tucker RM, Hamilton JR, Stevens DA: Concurrent bloodstream infection with Histoplasma capsulatum and Mycobacterium tuberculosis. J Med Vet Mycol 29:343, 1991 6. Carter JM, Green WR, Callender CO, et al: Pulmonary cavitation with Nocardia and Aspergillus in a renal transplant patient. .I Nat1 Med Assoc 82:527, 1990 7. Nwobu RA, Odugbemi T, Eneje RC, et al: Mycologic and bacteriologic investigations of clinical sputum specimens in Largos, Nigeria. West Afr J Med 8:274, 1989 8. Ratliff DP: Tuberculosis of the mandible. Br Dent J 135:122, 1973 9. Thilander H, Wennstrom A: Tuberculosis of the mouth and surrounding tissues. Oral Surg Oral Med Oral Path01 9:858, 1956 10. Heney C, Baise T, Cohen MA: Tuberculosis of the mandible: A case report. Pediatr Infect Dis .I 7:74, 1988 11. Worsaae N. Reibel J. Rechnitzer C: Tuberculous osteomvelitis of the mandible. Br J Oral Maxillofac Surg 22:93, 1984 12. Sachs SA, Eisenbud L: Tuberculous osteomyelitis of the mandible. J Oral Surg 44:425, 1977 13. Sepheriadou-Mavropoulou T, Yannoulopoulos A: Tuberculosis of the jaws. J Oral Maxillofac Surg 44:158, 1986 14. Jakhi SA, Parekh BK, Dadrevala FJ: Tuberculous osteomyelitis of me mandible: A case report. J Indian Dent Assoc 55:193, 1983 15. Wood RE, Housego T, Nortje CJ, et al: Tuberculous osteomyelitis in the mandible of a child. Pediatric Dentistry 9:317, 1987 16. Foster CF, Young WG: Tuberculous infection of a fractured mandible: Report of a case. J Oral Surg 28:686, 1970 17. Addlestone RB, Witt WS, Kaiser AB: Tuberculosis of the mandible oresentine as “lumuv iaw.” JAMA 241:2544. 1979 18. Khosla VM: Tuberculous o&myelitis of the mandible: Report of a case. J Oral Surg 28:848, 1970 19. Garber HT, Harrigan W: Tuberculous osteomyelitis of the mandible with a pathologic fracture. J Oral Sure. 36:144, 1978 20. Weidmann GM; MacGyegor AJ: Tuberculous-osteomyelitis of the mandible. Oral Sure Oral Med Oral Path01 28:632, 1969 21. Jones WC, Miller WE: Skeletal tuberculosis. South Med J 571964, 1963 22. Enarson DA, Ashley S, Grzybowski S, et al: Non-respiratory

RUGGIERO, HILTON,

23. 24. 25. 26. 27. 28. 29.

BRAUN

tuberculosis in Canada: Epidemiologic and bacteriologic features. Am J Epidemiol 112:341, 1980 Weir MR, Thornton GF: Extrapulmonary tuberculosis: Experience of a community hospital and a review of the literature. Am J Med 79:467, 1985 Alagumba L, Kekere-Ekun TA, Sawyer DR, et al: Primary tuberculous osteomyelitis of the mandible. J Maxillofac Surg 11:46, 1983 Nor GM: Tuberculous osteomyelitis of the mandible. Dent J Malaysia 10:48, 1988 Mishra YC, Bhoyar SC: Primary tuberculous osteomyelitis of the mandible: A case report. J Indian Dent Assoc 58:335, 1986 Davidson PT, Horowitz I: Skeletal tuberculosis: A review with patient presentations and discussion. Am J Med 48:77, 1970 Versfeld GA, Solomon A: A diagnostic approach to tuberculosis of bone and joints. J Bone Joint Surg Br 64:446, 1982 Halsey JP, Reeback JS, Barnes CG: A decade of skeletal tuberculosis. Ann Rheum Dis 41:7, 1982

1239 30. Newton P, Sharp J, Barnes KL: Bone and joint tuberculosis in Greater Manchester 1969-79. Ann Rheum Dis 41:1, 1982 31. Van Scoy RE, Wilkowske CJ: Antituberculous agents. Mayo Clin Proc 67:179, 1992 32. Goble M, Iseman MD, Madsen LA, et al: Treatment of 171 patients with pulmonary tuberculosis resistant to isoniazid and rifampin. N Engl JMed 328:527, 1993 33. Freidan TR, Sterlinn T. Pablos-Mendez A. et al: The emergence of drug-resistant-tuberculosis in New ‘York City. N l&g1 J Med 3281522, 1993 34. Glassroth J, Robins AG, Snider DE: Tuberculosis in the 1980’s. N Engl J Med 302:1441, 1980 35. McKenna MT, McCray E, Onorato I: The epidemiology of tuberculosis among foreign-born persons in the United States, 1986 to 1993. N Engl J Med 332:1071, 1995 36. Iseman MD, Starke J: Immigrants and tuberculosis control. N Engl J Med 332:1094, 1995 37. Iseman MD: Treatment of multidrug-resistant tuberculosis. N Engl J Med 329:784, 1993