Tuberculous osteomyelitis affecting periodontium: A rare case report.

Tuberculous lesions affecting periodontium are rare and seen as secondary infections localized to the soft tissues. With the advent of effective drug therapy, tuberculous lesions of the oral cavity have become rare. Involvement of the periodontium has seldomly been reported in the recent literature. We report a case of tuberculous osteomyelitis of mandible affecting periodontium leading to gingival recession and bone exposure in the mandibular premolar region in a 42-year-old female patient. The diagnosis was based on patient's medical and dental history, bacterial culture, clinical and radiographic examination, blood investigation, immunologic tests, histopathologic examination of the tissue specimen. Patient was already taking antitubercular chemotherapy prescribed by physician. Sequestrectomy and decortications were carried out to remove the affected bone. Healing was uneventful and there was no recurrence after 1½ year of follow-up. Antitubercular chemotherapy along with sequestrectomy and decortication are the treatment of choice for tuberculous osteomyelitic lesions affecting periodontium.

INTRODUCTION

Tuberculosis is a chronic granulomatous disease caused by Mycobacterium tuberculosis; it can affect any part of the body including oral cavity and periodontium. The average annual risk of tuberculosis infection is highest in sub-Saharan Africa (1.5–2.5%) and Asia (1.0–2.0%).[1] In comparison, the average annual risk in developed countries like Netherlands was estimated to be 0.012%.[2] Extra pulmonary tuberculosis is rare, occurring in 10–15% of all cases.[3] Tuberculosis shows a high incidence of coexistence with various immunocompromised conditions. In sub-Saharan Africa, about 80% of subjects with tuberculosis are coinfected with HIV, and it is estimated that in South Africa 30% of HIV-seropositive subjects have active tuberculosis.[4]

Tuberculous lesions of the oral cavity are so rare that this manifestation of the disease is often overlooked.[56] Oral tuberculosis can be primary or secondary. Primary oral tuberculous lesions are extremely rare and generally occur in young adults with associated caseation of the dependent lymph nodes; the lesion itself remains painless in most cases.[78] Oral tuberculosis accounts for up to 1.33% of HIV-associated opportunistic infections.[9] In contrast, secondary oral tuberculosis is seen in about 0.05–1.5% of reported cases and usually occurs in older adults.[1011] In oral tuberculosis, the most commonly affected site is the tongue; other sites include the lip, cheek, soft palate, uvula, gingiva and alveolar mucosa.[12] The lesions are seen as superficial ulcers,[1314] patches,[6] indurated soft tissue lesions[15] or even lesions within the jaw in the form of tuberculous osteomyelitis.[16] Tuberculous osteomyelitis is quite rare and constitutes <2% of skeletal tuberculosis, jaw involvement is even rarer.[17]

When oral lesions are the sole manifestation of tuberculosis, the clinician may face a diagnostic challenge. So, here, we report a rare case of tuberculous osteomyelitis affecting the periodontium in the form of gingival recession along with exposure of the affected bone.

CASE REPORT

A 42-year-old female reported to the Department of Periodontics and Oral Implantology, Pacific Dental College and Hospital, Udaipur with the chief complaint of dull pain, receded gums and feeling of roughness in inner aspect of lower right back teeth region since last 4 months.

Past medical and dental history

Patient gave a history of swelling in submandibular region from the past 5 months, along with the history of evening rise in temperature and weakness. The patient also had history of loss of appetite and weight loss. Patient also gave a history of dental trauma during root canal treatment and extraction in mandibular anterior teeth region just 2 days before the appearance of swelling in the submandibular region. Because of the swelling and elevated body temperature patient was referred to the physician, there fine needle aspiration cytology was carried out for the same. Pus was aspirated for the bacteriological investigation. In the culture of the pus, numerous colonies of Mycobacterium tuberculosis were identified. Along with that, the physician observed palpable and tender submandibular lymph nodes. So, on the basis of clinical and cytological examination Tuberculous Lymphadenitis was diagnosed.

DRUG HISTORY

Patient's drug history revealed that antituberculour drug isoniazid, rifampicin, pyrazinamide and ethambutol were initiated for initial 2 months followed by isoniazid and rifampicin for the following 4 months by physician. At the time of patient reported to the department, patient was already diagnosed as tuberculous lymphadenitis and was taking isoniazid and rifampicin once daily.

Extraoral examination

There was no extraoral swelling. Cervical and submandibular lymph nodes were not palpable.

Intraoral examination

Clinically, intraoral examination revealed that there was a gingival recession and bone exposure on the buccal and lingual aspect of the right side mandibular premolar region [Figure 1]. Grade 1 mobility was noticed on the mandibular right first premolar. Vitality test of both first and second premolar was positive.

Figure 1

Preoperative view shows gingival recession and bone exposure in premolar region

Radiographic examination

Radiographic examination showed osteomyelitic changes with bone loss along the middle third of the root surface [Figure 2]. Chest radiograph revealed no abnormalities.

Figure 2

Preoperative radiograph shows osteomyelitic changes in between premolars

Hematological tests

Here the values were within normal limits, except for a marginal rise in leukocyte count (12.8 × 108/L) and an elevated erythrocyte sedimentation rate (ESR) of 58 mm/h, which raised the possibility of one of the commoner causes of high ESR, TB.

Mantoux test

The tuberculin (Mantoux) test was positive, suggesting tubercular infection.

Microbiologic testing

A culture of the sputum, obtained by forceful coughing, was negative for M. tuberculosis.

Immunologic testing

An immunologic test to detect antibodies against Mycobacterium in the patient's serum (ELISA) was positive. Polymerase chain reaction (PCR) assay was also carried out, positive PCR results confirmed the presence of M. tuberculosis in the tissue samples.

Case management

After physician's consent and finishing the 6 months regimen of anti-tubercular therapy sequestrectomy and decortications of the affected bone was planned. Scaling and root planning were performed as a part of phase I therapy. Surgical intervention was planned after 3 weeks of the phase I therapy. Under local anesthesia, bucally and lingually sulcular incisions were placed. After reflection of the mucoperiosteal flap, a sequestrum of 1.5 cm × 2.5 cm surrounded by granulation tissue was seen. Sequestrectomy was performed with surgical burs and Schluger's bone file [Figure 3]. Difference between healthy bone and sequestrum was clearly appreciated as sequestrum doesn’t bleed on injury [Figure 4]. Bone was excised until normal bleeding bone was encountered. All granulation tissue was removed, and root planning was performed. Interrupted sutures were placed, and periodontal pack was applied. After 7 days sutures were removed. Healing was uneventful.

Figure 3

Sequestrum removed with the help of bone file

Figure 4

Sequestrum removed until the healthy bleeding bone encountered

Bony tissue was obtained for the histological examination [Figure 5]. Histological examination shows numerous bony trabeculae with the absence of peripheral osteoblastic rimming and osteocytes within the lacunae with the presence of ragged or irregular borders representing sequestrum. Focal granulomatous process with Langerhan's giant cells and epitheloid cells were found in adjacent marrow cavities [Figures 6 and 7]. This histological examination was suggestive of tuberculous osteomyelitis.

Figure 5

Sequestrum removed for histopathological examination

Figure 6

Histologic examination shows osteomyelitic changes in the bone and focal granulamatous process with langerhans's giant cells (in arrow)

Figure 7

Close view (×10) of focal granulamatous process with Langerhan's giant cells

Final diagnosis

In view of these findings, final diagnosis tuberculous osteomyelitis was made.

Clinical outcome

Patient was recalled for regular follow-up. After 1½ year of follow-up, there was no recurrence of lesion [Figures 8 and 9].

Figure 8

Postoperative view after 1½ year of follow-up. There is no sign of recurrence

Figure 9

Radiograph after 1½ year follow-up

DISCUSSION

In the Indian population, the average prevalence of all forms of tuberculosis has been reported to be 5.05/1000.[18] Compared with tuberculous involvement of other parts of the body, occurrence of this disease in the oral cavity and jaw bones is relatively rare. As a consequence, clinicians are not sensitized to the disease as a part of a differential diagnosis, and there are undoubtedly patients in whom the correct diagnosis and therapy are delayed or missed.

The mechanism of primary inoculation into the oral mucous membrane is not clearly understood. One reason for the rare occurrence of tuberculosis of the gingiva may be that the intact squamous epithelium of the oral cavity resists direct penetration by bacilli.[13] This resistance has been attributed to the thickness of the oral epithelium, the cleansing action of saliva, local pH and antibodies in saliva.[3] Even if the onset of infection is by hematogenous spread, injured or inflamed tissue tends to localize bloodborne bacteria. However, the mode of entry of the organism may be through a break in the mucous membrane caused by local trauma.[19] Where the infection involves bone, the mode of entry is thought to be through an extraction socket. However, there is general consensus that secondary tuberculosis spreads by a hematogenous route.[20]

There is a high incidence of tuberculosis in immunocompromised patients as well, especially in HIV patients such that HIV and tuberculosis coinfection constitutes a serious public health problem. It is estimated that one-third of the persons living with HIV infection are coinfected with tuberculosis. This coinfection is so clinically relevant that current guidelines recommend that HIV testing be performed in every individual diagnosed with tuberculosis.[21]

With the rarity of such lesions in periodontium, its diagnosis may be difficult and often missed in cases of delayed or absence of pulmonary and other systemic signs. In the present case report, Tuberculous Lymphadenitis was already diagnosed before patient reported to the department. Diagnosis was reconfirmed by immunologic test. The osteomyelitic changes of the periodontium should be considered a tuberculous lesion since tubercle bacilli were cultured from abscess cavity and epithoiloid cell granuloma was found in the affected bone. Pain, sequestration and an aggressive course of the disease are characteristic clinical findings of tuberculous osteomyelitis.

In the present case, the patient was already prescribed antitubercular regimen by the physician before 4 months, but there was no sign of regression in the lesion. Therefore, sequestrectomy and decortications were carried out to remove the osteomyelitic lesion.

SUMMARY

Tuberculous lesions of the oral cavity can assume a nonspecific clinical appearance. When jaw bones are involved, the disease presents features of chronic osteomyelitis. In this assessment, a complete physical examination should also be included, with diagnostic tests such as chest radiographs, biopsy specimens for histological studies and culture of the organism. An early diagnosis with prompt treatment will usually result in a complete cure.