Reactive granulomatous lesion in the pterygomandibular fossa mimicking a temporomandibular disorder

Reactive granulomatous lesion in the pterygomandibular fossa mimicking a temporomandibular disorder

J Oral Maxillofac Surg 60:1203-1206, 2002 Reactive Granulomatous Lesion in the Pterygomandibular Fossa Mimicking a Temporomandibular Disorder Andre´s...

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J Oral Maxillofac Surg 60:1203-1206, 2002

Reactive Granulomatous Lesion in the Pterygomandibular Fossa Mimicking a Temporomandibular Disorder Andre´s F. Herrera, DDS,* Louis G. Mercuri, DDS, MS,† and Maria M. Picken, MD, PhD‡ The diagnosis of reactive granulomatous lesions in the head and neck region can challenge the oral and maxillofacial surgeon because their presentation may mimic other pathological processes. We report the case of a patient with a reactive granulomatous lesion in an anatomically obscure area, the pterygomandibular fossa. The patient presented with signs and symptoms of a temporomandibular joint disorder.

Report of a Case A 39-year-old white man was referred for evaluation with a 5 month history of limitation of mouth opening. The patient also had an associated painful and slowly progressive swelling on the left side of his face. A review of systems was unremarkable and noncontributory. The patient’s past medical history was remarkable for hypercholesterolemia, which was being treated with Lipitor (Parke-Davis, a division of Warner-Lambert, Morris Plains, NJ). His social history was remarkable for smoking one pack of cigarettes per day for 15 years as well as for moderate alcohol consumption. The patient is a Polish immigrant who works as supervisor in a meat-processing factory. Physical examination revealed a hard, nontender swelling of the left parotid area. The skin in this area was normal in color and texture, and no sinus tracts were seen. The fifth and seventh cranial nerves were grossly intact bilaterally. No lymphadenopathy was noted. Interincisal opening was measured as 10 mm without deviation and was associated with a mild pain in the left preauricular area. Intraorally, a hard, nontender swelling of the left medial mandibular ramus was present. The mucosa covering this area appeared normal. The remaining partially edentulous dentition included multiple amalgam restorations in acceptable condition; no acute odontogenic infection was evident. The oropharynx was normal in size and appearance.

Received from Loyola University Medical Center, Maywood, IL. *Resident, Division of Oral and Maxillofacial Surgery. †Professor, Division of Oral and Maxillofacial Surgery and Dental Medicine. ‡Professor, Department of Pathology. Address correspondence to Dr Mercuri: Loyola University Medical Center, 2160 South First Avenue, Maywood, IL 60302; e-mail: [email protected] © 2002 American Association of Oral and Maxillofacial Surgeons

0278-2391/02/6010-0021$35.00/0 doi:10.1053/joms.2002.35035

A panoramic radiograph revealed a multilocular radiolucent lesion with ill-defined borders that extended from the left coronoid process to the superior aspect of the left mandibular angle. The mandibular cortex in this area appeared radiographically intact. No dental, apical, or periodontal lesions were found (Fig 1). A computed tomography (CT) scan including axial and coronal images of the facial skeleton revealed abnormal scalloping of the lingual cortex of the left mandibular ramus and erosion of the left lateral pterygoid plate (Fig 2). Multiple intraoral incisional biopsies and a fine needle aspiration (FNA) suggested a low-grade spindle cell neoplasm rather than a reactive process. The results of these biopsies were nonconclusive except for the presence of focal granuloma. At this point, the working differential diagnosis included lymphoma, sarcoidosis, rhabdomyosarcoma, low-grade fibrosarcoma, granulomatous reaction consistent with Mycobacterium tuberculosis, actinomycosis, or fungal infection. A facial magnetic resonance image (MRI) revealed a prominence of the left medial pterygoid, left temporalis, and masseter muscles (Fig 3). The MRI was suggestive of myositis or a mass arising from the left medial pterygoid muscle extending to the skull base. Because the lesion was localized medial to the mandible and prior intraoral biopsies were nonspecific, the patient was taken to surgery. Under general anesthesia, an incisional biopsy was performed using a submandibular approach. A large, protruding soft tissue mass was visualized arising from the medial aspect of the left mandible (Fig 4). The medial pterygoid muscle was medially displaced by the mass. The mass was indurated, with a bosselated appearance, and tenuously attached to the bone. Part of the lesion was sent for frozen section; the results were negative for malignancy but suggestive of a granulomatous lesion. Other tissue samples underwent routine section as well as microbiology and tissue culture. The specimens included several lymph nodes and the left coronoid process. The permanent sections showed connective tissue with multiple necrotizing granulomas (Fig 5); the lymph nodes were histologically normal. No evidence of malignancy was found in any of the samples. All the specimens were negative for acid-fast bacilli and fungi. The anaerobic and aerobic cultures reported few colonies of normal oral flora. No anaerobic micro-organisms or Actynomyces species were isolated after a 2-week period. In addition, no vasculitis was seen histologically. Purified protein derivative test was positive; however, the patient had reported being vaccinated with Bacille Calmette-Gue´rin (BCG) in Poland during his childhood. A chest radiograph showed all the pulmonary fields to be clear. Based on the patient’s mandibular functional limitations, the decision was made to excise the entire lesion. Because of the lesion’s location at the pterygomandibular fossa and

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REACTIVE GRANULOMATOUS LESION MIMICKING TMD

FIGURE 1. Panoramic radiograph showing the multilocular, radiolucent pattern of the lesion in the left mandible.

its proximity to the skull base, the surgical team was extended to include also a skull base surgeon. At surgery, a left hemicoronal incision was made to expose the left infratemporal fossa. A submandibular approach was used to expose the left mandibular body, angle, and ramus. A 2-mm, 6-hole mandibular plate was contoured to the lateral mandibular cortex. A vertical ramus osteotomy was performed to expose the lesion. The superior portion of the mass extended up to the medial pole of the condyle. The entire mass was dissected free, allowing the delivery of the mass through the pterygomandibular space. Hemostasis was achieved, and the mandible was fixed with the preformed osteosynthesis plate. The specimen was divided into several samples which were sent for routine histopathological evaluation as well as microbiology examination and culture. The patient was discharged after an uneventful postoperative period of 3 days. Physical therapy using the Therabite (Therabite Corporation, West Chester, PA) device was started 1 week after surgery. The final pathological examination revealed soft tissue, skeletal muscle, and peripheral nerves with necrotizing granulomas. All the special stains were negative for micro-organisms and vasculitis. Microbiological examination

FIGURE 3. Axial MRI. The left medial pterygoid appears enlarged compared with the contralateral counterpart.

showed few colonies of Streptococcus viridans and pneumonie as well as few colonies of coagulase-negative Staphylococcus species. No Actinomyces species were isolated. Two months later, the cultures remained negative for fungi or acid-fast bacilli. Ten months after the last surgical procedure, the patient presented with an asymptomatic interincisal opening of 36 mm without deviation. The occlusion was stable and consistent with the preoperative condition.

Discussion This case presented several difficulties in terms of diagnosis and management. Restriction of mandibular opening, as a unique complaint on presentation, can be caused by multiple disorders, ranging from mus-

FIGURE 2. CT scan, coronal image. Notice the lingual cortex scalloping in the left ascending ramus of the mandible.

FIGURE 4. Clinical photograph shows the protrusion of the lesion from the medial aspect of the left mandible.

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FIGURE 5. The paraffin sections show multiple well-formed granulomas that demonstrated focal necrosis (insert).

cular dysfunction to intra-articular or extra-articular pathology (Table 1).1 In this particular case, after ruling out intracapsular pathology, attention was directed to the extra-articular tissues (fibrous, muscular, osseous, adipose connective tissues, and peripheral nerves). A thorough evaluation revealed the presence of a mass in the left pterygomandibular fossa; the differential diagnosis included neoplasm and infectious and autoimmune processes. Among the neoplastic processes, non-Hodgkin’s lymphoma (NHL) was very high in the differential diagnosis. About 24% of NHL cases present in extranodal sites. Among the extranodal NHL cases, 25% present in the head and neck. Two thirds of the head and neck cases are located in the Waldeyer’s ring because it represents the largest concentration of lymphoid tissue in this region.2-4 Although the FNA results were suggestive for this diagnosis, the lack of systemic signs and symptoms did not support it. Moreover, non-Hodgkin’s lymphoma is typically confined to the lymph nodes and presents less frequently as an extranodal lesion in the soft or hard tissues. There was also a high suspicion for a malignant neoplasm because of the clinical and radiographic findings, but all biopsies proved negative for malignancy. Because the histopathological studies showed a necrotizing granulomatous process, the diagnosis of extrapulmonary tuberculosis was also high on the differential diagnosis list. Extrapulmonary tuberculosis accounts for approximately 16.2% of all the diagnosed tuberculosis cases.5,6 The oral cavity is rarely involved in cases of extrapulmonary tuberculosis (1.4%). In those cases, soft tissue compromise is more common than bone involvement. However, when the facial skeleton is involved, the mandible is effected more often than the maxilla. Diagnostic tests such as PPD were of no value for this patient because of his history of BCG vaccination. His chest radiograph was nega-

tive. More significantly, the specimens were negative for AFB, and special stains and cultures were negative for M tuberculosis. Actinomycosis can be associated with necrotizing granulomas in the head and neck region, and account for more than 50% of such cases in the body. This gram-positive bacterial infection presents clinically as a chronic suppurative infection. The discharge obtained from sinus tracts may contain a solid yellow material (sulfur granules) containing colonies of these bacteria. The diagnosis is made by culture, direct demonstration from biopsied material, or flouresceinconjugated antiserum.7 In this case, neither the clinical presentation nor the microbiological studies were positive for actinomycosis. The absence of other organ involvement and systemic signs and symptoms suggested that sarcoidosis was not the cause of this granulomatous process in the pterygomandibular region.8 Moreover, sarcoidosis is typically associated with non-necrotizing granulomas. After ruling out any neoplastic, infectious, autoimmune, or inflammatory process as causing the lesion, and without a history of trauma or odontogenic infection, the only episode that was coincident with the onset of symptoms was the patient’s visit to his general dentist for restorative treatment of the maxillary left second molar. During this visit, the patient received a posterior superior alveolar nerve block (PSA) with local anesthesia. This isolated fact lead us to

Table 1. CAUSES OF RESTRICTED MANDIBULAR OPENING

Intra-Articular

Extra-Articular

Trauma Hemarthrosis Fracture Birth injury Dislocated meniscus Infection Arthritic Odontogenic Otogenic

Soft tissue obstruction Infection Edema Scar tissue Neoplasia Radiation therapy Foreign bodies Congenital malformations Hecht, Beals, and Wilson syndrome Bone obstruction Enlarged coronoid process Fractured, displaced zygoma Zygomatic arch fracture Elongated styloid process Jacob’s disease (exostoses at posterior aspect of zygoma) Neurologic disorders Tetanus Hysteria Cerebral lesions Extrapyramidal reactions

Hematogenic Ankylosis

Reprinted with permission.1

1206 hypothesize that pathogenic bacteria were introduced to the infratemporal fossa and ultimately the pterygomandibular space during a PSA block leading to the development of a chronic abscess. The lack of signs or symptoms related to the infectious process may be attributable to the patient’s physical status. The patient is a well-nourished, well-developed man with an unremarkable medical history. Those factors can influence the development of a subacute infection that succeeded within the aponeurotic spaces posteriorly and underwent granulomatous transformation. This hypothesis can be supported by Hopps’9 formula: Severity of infection ⫽

(Number of microorganisms) ⫻ (virulence of organisms) Resistance of host



Portal of entry

By applying this formula to the present case, one can assume that the severity of the infection in this case was attenuated by an immunocompetent host and low-virulent bacterial strains in small number. Multiple cases of the development of infratemporal fossa or pterygomandibular space abscesses after a PSA block have been reported.10 In those cases, one

of the cardinal signs on presentation is trismus, as well as swelling at the level of the mandibular angle.11

References 1. Mercuri LG: The Hecht, Beals, and Wilson syndrome: Report of case. J Oral Surg 39:53, 1981 2. Piatelli A, Croce A, Tete S, et al: Primary non-Hodgkin’s lymphoma of the mandible: A case report. J Oral Maxillofac Surg 55:1162, 1997 3. Miller RI: Non-Hodgkin’s lymphoma of the lip: A case report. J Oral Maxillofac Surg 51:420, 1993 4. Rooney N, Ramsay AD: Lymphomas of the head and neck: The B-cell lymphomas. Oral Oncol Eur J Cancer 30B:155, 1995 5. Murray JF, Nadel JA: Textbook of Respiratory Medicine. Philadelphia, PA, Saunders, 1994, pp 1094-1160 6. Florio S, Ellis E, Frost DE: Persistent submandibular swelling after tooth extraction. J Oral Maxillofac Surg 55:390, 1997 7. Neville B, Damm DD, Allen CM, et al: Oral & Maxillofacial Pathology. Philadelphia, PA, Saunders, 1995, 156-157 8. Newman LS, Rose CS, Maier LA: Medical progress: Sarcoidosis. N Engl J Med 336:1224, 1997 9. Hopps HC: Principles of Pathology. New York, NY, AppletonCentury-Crofts, 1964, p 204 10. Kitay D, Ferraro N, Sonis ST: Lateral pharyngeal space abscess as a consequence of regional anesthesia. J Am Dent Assoc 122:56, 1991 11. Cohen SG, Quinn PD: Facial trismus and myofascial pain associated with infections and malignant disease: Report of five cases. Oral Surg Oral Med Oral Pathol 65:538, 1988