Tuberculous Osteomyelitis of the Mandible With Diffuse Swelling of the Floor of the Mouth: A Case Report

CASE REPORT

Tuberculous Osteomyelitis of the Mandible With Diffuse Swelling of the Floor of the Mouth: A Case Report Shuang Bai, MDS, DDS,* and Chang-Fu Sun, MD, DDS, PHDy Primary orofacial tuberculosis (TB) is uncommon, especially with regard to the jaw. We report an unusual case for which the final diagnosis was tuberculous osteomyelitis of the mandible with cervical tuberculous lymphadenitis. The follow-up examinations for our patient showed complete regression of the swelling and healing of the mandibular lesion after 4 months of TB antibiotic therapy. The purpose of the present study was to alert clinicians to our findings and encourage them to consider oral TB in the differential diagnosis for jaw lesions with multiple enlarged cervical lymph nodes. Crown Copyright Ó 2014 Published by Elsevier Inc on behalf of the American Association of Oral and Maxillofacial Surgeons. All rights reserved J Oral Maxillofac Surg 72:749.e1-749.e6, 2014 Tuberculosis (TB) is a chronic systemic disease caused by various strains of mycobacterium, usually Mycobacterium tuberculosis, although M. avium, M. bovis, M. kansasii, and M. scrofulaceum have also been linked to the disease.1 Owing to the protective effect of an intact oral mucosa, oral intrinsic saprophytic bacteria, the salivary cleaning action, and salivary protease digestion, primary orofacial TB is rare, and few cases have been reported. This has been especially true when no evidence of pulmonary disease or systemic spread has been found. The incidence of extrapulmonary TB has shown an increase in recent years. Extrapulmonary infections, without any systemic involvement, can present as an oral mucosa ulcer, granuloma, salivary gland involvement, orofacial TB, tuberculous lymphadenitis, or tuberculous osteomyelitis of the jaws.2,3 Tuberculous osteomyelitis of the mandible has accounted for less than 2% of all skeletal TB.4 Because the clinical manifestation of TB is highly variable and published data are lacking on oral TB, it has quite often been misdiagnosed. We have reported a case of tuberculous osteomyelitis of the mandible with cervical tuberculous lymph-

adenitis in a young man that had masqueraded as malignancy. We sought to promote discussion of the diagnostic modalities and management and to encourage clinicians to consider TB in the differential diagnosis.

Case Report A 31-year-old man was initially referred to the outpatient department in our center. He was subsequently admitted to the head and neck surgery service with a suspected malignant neoplasm of the sublingual gland for additional evaluation and management. The patient complained of swelling on the floor of the mouth and in the submandibular region, in addition to pain on swallowing, which had persisted for 2 months. One week earlier, the patient had felt numbness in the lower lip on the left side. The patient was a manual worker. The general examination revealed that he was moderately built and well nourished and had experienced no systemic symptoms, such as fever, cough, hemoptysis, or weight loss or anorexia, during the past 2 months. However, he

Received from Departments of Oromaxillofacial-Head and Neck

trict, Shenyang, Liaoning 110002, People’s Republic of China;

Surgery and Oral and Maxillofacial Surgery, China Medical

e-mail: [email protected]

University School of Stomatology, Shenyang, Liaoning, People’s

Received October 16 2013

Republic of China.

Accepted November 28 2013 Crown Copyright Ó 2014 Published by Elsevier Inc on behalf of the American

*Resident. yProfessor, Head of Department of Oromaxillofacial-Head and Neck Surgery. Address correspondence and reprint requests to Dr Sun: Depart-

Association of Oral and Maxillofacial Surgeons. All rights reserved 0278-2391/13/01498-5$36.00/0 http://dx.doi.org/10.1016/j.joms.2013.11.030

ment of Oral and Maxillofacial Surgery, China Medical University School of Stomatology, No. 117, Nanjing North Street, Heping Dis-

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reported experiencing weakness, fatigue, and night sweats 2 weeks before the examination. He was a cigarette smoker, with a 5 pack-year history. He was allergic to penicillin and had no history of contact with suspicious allergens. He also had no history of any trauma to the orofacial region. Extraorally, a mild swelling was observed in the submandibular region. The overlying skin had a normal appearance and temperature. On palpation, the bilateral submandibular lymph nodes were enlarged and movable. Intraorally, his oral hygiene was not optimal. The bimanual examination of the floor of the mouth revealed a firm, tender swelling mainly involving the anterior region of the floor of the mouth. The overlying mucosa of the anterior floor of the mouth was intact and normal, with no evidence of ulcer. The mandibular right lateral incisor and canine teeth were mobile. Suspecting a malignant neoplasm of the sublingual gland, the patient was referred to the radiology department at our center for additional examinations. The digital panoramic radiograph showed an ill-defined and radiolucent area in the anterior mandibular region, especially in the apical region of the right anterior teeth. Mesioangular impaction of the lower third molars was also revealed. A contrast-enhanced computed tomography scan was taken to determine the extent of the lesion. In the bone window, the findings revealed erosion of the anterior mandible and absorption of the cortical plate. In the soft tissue window, a suspicious space-occupying lesion was present, with peripheral enhancement on the floor of the mouth. Multiple enlarged, bilateral, cervical lymph nodes (Fig 1) were visualized. Magnetic resonance imag-

TUBERCULOUS OSTEOMYELITIS OF THE MANDIBLE

ing of the floor of the mouth demonstrated long T2-weighted signals and multiple, unequally sized, enlarged lymph nodes surrounding the bilateral submandibular glands and carotid sheaths (Fig 2). Because of these findings, ultrasonography of the cervical region was performed, revealing multiple cervical lymph nodes of increased diameter (#2.5  0.9 cm on the right and 2.9  0.9 cm on the left). However, no sign of any suspicious masses was seen. With approval from the patient, an incisional biopsy was performed to obtain a specimen of the swelling tissue on the floor of the mouth. The histopathologic examination showed epithelioid histiocytes and multinucleated giant cells surrounded by a lymphocytic infiltrate (Fig 3). From this finding, a granulomatous infection was provisionally diagnosed. A microscopic examination was performed and revealed no foreign materials. Acid-fast staining was performed, with negative findings. Because no evidence of an ulcer in the patient’s oral cavity or of suppurative lymphadenitis was found, Crohn’s disease and chronic granulomatous disease were also excluded from the diagnosis. Given the prevalence of TB in northeast China, primary orofacial TB was suspected. Additional investigations to confirm TB were advised. Hematologic examination revealed an obvious increased erythrocyte sedimentation rate (28 mm/hour) and increased mononuclear leukocytes. The patient showed a strongly positive (21 mm) reaction to the purified protein derivative (PPD) test (Fig. 4). This result also ruled out the diagnosis of sarcoidosis. An enzyme-linked immunosorbent assay test for human immunodeficiency virus (HIV)

FIGURE 1. Axial computed tomography scan with contrast in the bone window showing erosion of the anterior mandible and absorption of the cortical plate (white arrow). Bai and Sun. Tuberculous Osteomyelitis of the Mandible. J Oral Maxillofac Surg 2014.

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FIGURE 2. Coronal magnetic resonance imaging scan showing long T2-weighted signals on the floor of the mouth (white arrow). Bai and Sun. Tuberculous Osteomyelitis of the Mandible. J Oral Maxillofac Surg 2014.

types 1 and 2 was negative. The chest radiograph showed no evidence of pulmonary TB. Five days after being admitted to the hospital, the patient complained of a fistula on his anterior mandibular gingiva. With the patient under local anesthesia, we performed curettage of the fistula and obtained yellowish-white secretions for additional examination. Finally, we used the polymerase chain reaction (PCR) to confirm the presence of the M. tuberculosis complex. We also performed fungal culture experiments, which took approximately 4 weeks. Eventually, a negative result excluded a fungal infection. From these results, we finally diagnosed tuberculous osteomyelitis of the mandible with tuberculous lymphadenitis of the neck.

The patient was transferred to the respiratory medical department for TB antibiotic therapy. The regimen consisted of isoniazid, rifampicin, pyrazinamide, and ethambutol for the initial 2 months, followed by isoniazid and rifampicin for the next 4 months. Because of the side effects associated with antibacterial TB drugs, regular tests of the hepatic enzymes, kidney function, and blood were performed during the treatment course. Four weeks later, the patient had exhibited complete regression of the swelling and symptomatic relief. The fistula on the anterior mandibular gingiva had healed after 6 weeks. At the follow-up examination 4 months later, the panoramic radiograph

FIGURE 3. Photomicrograph (hematoxylin and eosin stain, original magnification 10) showing epithelioid histiocytes, multinucleated giant cells (black arrow), surrounded by a lymphocytic infiltrate. Bai and Sun. Tuberculous Osteomyelitis of the Mandible. J Oral Maxillofac Surg 2014.

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FIGURE 4. Result of purified protein derivative test showing an induration and size greater than 1 cm (black arrow). Bai and Sun. Tuberculous Osteomyelitis of the Mandible. J Oral Maxillofac Surg 2014.

showed that the lesion in the mandible had also healed (Fig 5).

Discussion TB is an ancient infectious disease that still threatens lives worldwide. The World Health Organization (WHO) reported that an estimated 8.6 million people had developed TB and 1.3 million had died of the disease (including 320,000 deaths among HIV-positive people) in 2012.5 Approximately 3 million people who developed TB in 2012 were missed by national notification systems.5 The prevalence of TB has resulted from a range of factors, including a rapidly increasing population, poor socioeconomic conditions, increasing HIV infection rates, and development of multidrug-resistant bacterial strains. Primary oral TB accounts for 0.1 to 5% of all TB infections and are usually reported in younger patients.6 Orofacial TB lesions have been reported in the tongue,6,7 lip,8 cheek,9 parotid gland,10 and jaws.2 The initial symptoms of primary oral TB can include swelling, pain, teeth loosening, nonhealing ulcers, and fistula formation. In addition, cervical lymphadenopathy will usually present as discrete or matted masses. Our patient’s presenting symptoms did not concur with the symptoms observed in other patients. We observed swelling of the floor of the mouth and submandibular region in conjunction with a mycobacterial infection. Our final diagnosis for our patient was tuberculous osteomyelitis of the mandible with tuber-

culous lymphadenitis. This is very rare, and, thus, we believe our case requires special attention. The radiographic examinations can show the extent of the lesion and will usually show a diffuse and blurring radiolucency, erosion of the cortical plates, and, even, a mixed radiolucent-radiopaque appearance in some cases.2 In the present case, our findings concurred with those from other published tuberculous osteomyelitis. Ultrasound imaging of the cervical masses is essential to distinguish solid from cystic masses. According to the published data, lymphadenitis (scrofula) is one of the most common extrapulmonary manifestations of TB.11 In the present case, the patient had multiple enlarged cervical lymph nodes, which are associated with lymphadenitis, indicating the presence of a microbial infection. Histologically, TB can present as a caseous necrotic granuloma with giant cells; however, this was not observed in our patient. This could have been because it is difficult to perform a biopsy on diffuse swelling tissue. Fine needle aspiration cytology of the lymph nodes can achieve the final diagnosis without open biopsy; however, the smears will always show a low positive rate for acid-fast bacilli using the ZiehlNeelsen stain.12 Hence, the low efficiency of the histologic examination has made it difficult to focus on the definite diagnosis in primary oral TB cases. Clinicians in developing countries are acutely aware of the possibility of TB infection owing to the prevalence of the disease in the developing world. Once TB has been suspected, chest radiographs are necessary to rule out pulmonary TB. In our patient, we

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FIGURE 5. Top, Panoramic radiograph showing the lesion in the mandible (white arrows), and Bottom, 4-month follow-up panoramic radiograph showing evidence of the healing of the lesion. Bai and Sun. Tuberculous Osteomyelitis of the Mandible. J Oral Maxillofac Surg 2014.

did not find enough evidence to diagnose pulmonary TB. In addition, a routine PPD test should be performed, although a positive reaction can be affected by bacille Calmette-Guerin vaccination.

With the help of numerous atypical clinical examination findings and the PCR test, we determined the final diagnosis for the present patient was tuberculous osteomyelitis of the mandible with tuberculous

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lymphadenitis. A study by Karbach et al9 reported that histopathologic morphology followed by nested PCR is the key to the diagnosis of ambiguous cases. We, therefore, encourage clinicians to apply advanced molecular biologic methods when attempting to diagnose awkward cases. In accordance with the WHO guidelines13 regarding standard regimens for new patients with TB, our patient received a multidrug regimen that included isoniazid, rifampicin, pyrazinamide, and ethambutol for the initial 2 months, followed by isoniazid and rifampicin for the next 4 months. Although a previous report by Sharma et al14 suggested that aggressive debridement with primary closure is essential to assist chemotherapy and prevent recurrence, we found this was not necessary. Our patient recovered completely from the swelling and the lesion in the mandible after chemotherapy, and performing an excision was not necessary. In the present case, we encountered a series of unusual results when diagnosing primary orofacial TB. TB can manifest in a variety of ways, and patients can present with a range of different symptoms, making the disease difficult to diagnose. Therefore, we suggest that clinicians consider TB in the differential diagnosis when patients present with orofacial masses and jaw lesions. An early diagnosis and determining the appropriate treatment is crucial in the treatment of TB.

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