Abstract
Maxillofacial tuberculosis (TB) is rare. The cases of 19 patients showing extreme bony destruction in the mandible, collected over a 3-month period in West Africa, are presented. Clinical, radiographic, and histological evidence indicated Mycobacterium tuberculosis as a possible cause. Further studies are in progress.
The authors have previously reported 60 cases of extreme osteonecrosis of the jaw (ONJ) based on sporadically collected records over a 3-year period from the hospital ship m/v Africa Mercy, while it was serving in Liberia, Benin, and Togo in West Africa. Many of the patients showed unilateral facial swelling, persistent sinus tracts, and destruction of the jaw bone. The degree of bony destruction was similar to that reported previously for conditions such as phossy or radium jaw. However, the pattern of destruction seen in the group of 60 patients was not the same in all patients. Some patients had maxillary lesions, although the preponderance was for mandibular cases. Furthermore, it seemed that the osteonecrosis was of two different types. One type of defect produced extremely necrotic ‘greyish’ bone sequestra of the type associated with ‘phossy jaw’. This manifestation occurred in both the maxilla and mandible. Patients with this type of necrosis did not have gross swelling and sinus tracts. The second type, by far the most common in the cohort of 60 cases, was characterized by unilateral gross facial swelling, frequently with a draining fistula, either intraorally or extraorally.
In this study, the association between tuberculosis (TB) and the development of necrosis was examined in the mandibular samples of 19 additional patients from Liberia and Sierra Leone. The samples were collected over a 3-month period from outpatients who voluntarily attended the Mercy Ships Dental Clinic while the ship Africa Mercy was stationed in Sierra Leone for a period of 9 months and from outpatients voluntarily attending Trinity Dental Clinic in Liberia. The osteonecrosis of these additional patients strongly resembled the second type of disease process described for the first group of 60 patients, both clinically and in the pattern of bony destruction of the mandible.
A potential link between TB and destruction of the jaw bones is suggested by three recent case reports describing patients with a tuberculous lesion in the condylar head of the mandible. In one case, a bone scan demonstrated an inflammatory destructive process in the condyle, ascending ramus, and angle. This pattern of destruction has a notable resemblance to the anatomical location of the ONJ in the 19 patients included in the current study. The second and third case reports presented data in support of the conclusion that the condylar head lesion was a primary tuberculous lesion.
Methods
Patient population
The tissue samples of the 19 participants in this study were collected between March and June of 2011 from patients in Sierra Leone ( n = 17) and Liberia ( n = 2). The patients were voluntarily attending dental clinics for the treatment of pain and swelling in the facial region. All of the patients presented with unilateral gross facial swelling, frequently with a draining fistula, either intraorally or extraorally. The patients answered a brief questionnaire regarding their general health and occupation. They were subsequently photographed to document the clinical appearance at the time of presentation.
Gross bony destruction was demonstrated by panoramic radiography and/or cone beam computed tomography (CBCT) in all but one of the cases. Facilities and logistics at the clinics limited the possibilities of chest X-rays and blood tests.
The patients all underwent a surgical debridement procedure as part of their treatment. After their surgical procedure was complete, and subsequent to obtaining informed consent from the patient, the discarded surgical debris was collected in 10% formaldehyde.
Specimen analysis
Micro-computed tomography (micro-CT)
High-resolution three-dimensional (3D) micro-CT scans (MicroXCT-200; Zeiss) were used to evaluate the degree and pattern of bony destruction in the condylar head of samples from four patients.
Histology and immunohistochemistry of soft tissue samples
Based on the limited amounts of soft tissue associated with the bony fragments, samples from four patients were selected for paraffin embedding, sectioning, and haematoxylin and eosin (H&E) staining and immunohistochemistry, following standard protocols. Sections of samples from two of the four patients were stained with antibodies against EMR-1 (PA5-33502; Thermo Fisher Scientific, MA, USA), a marker for mature macrophages. In addition, immunohistochemical staining with antibodies (GWB-EF714E; GenWay Biotech Inc., CA, USA) against purified proteins derived from Mycobacterium tuberculosis was used to determine whether mycobacterial proteins were present in the soft tissue samples.
Results
Clinical characteristics
Clinical characteristics are summarized in Table 1 . All 19 patients showed destructive lesions in the mandible.
| Patient | Sex | Age, years | Location | Specified location | Discharging extraoral sinus | Exposed bone intraorally | Additional signs and symptoms |
|---|---|---|---|---|---|---|---|
| 1 | M | 28 | Mandible | Bilateral angle/condyle | Yes | No | Multiple extraoral fistulae |
| 2 | F | 22 | Mandible | Left body | Yes | Yes | Large extraoral wound with necrosis |
| 3 | F | 20 | Mandible | Right ramus/condyle | No | No | Discharging intraoral wound |
| 4 | M | 56 | Mandible | Left body/angle/condyle | Yes | No | Multiple discharging extraoral fistulae |
| 5 | F | 24 | Mandible | Left body/angle/condyle | Yes | Yes | Discharging intraoral wound |
| 6 | F | 23 | Mandible | Left angle/condyle | Yes | No | Multiple discharging extraoral fistulae |
| 7 | F | 18 | Mandible | Left angle/condyle | No | No | Discharging intraoral wound |
| 8 | M | 32 | Mandible | Anterior body | No | No | Multiple mobile teeth |
| 9 | M | 41 | Mandible | Right ramus/coronoid | No | No | Closed extraoral sinus |
| 10 | M | 23 | Mandible | Left angle/ramus/condyle | No | No | |
| 11 | F | 70 | Mandible | Left body | No | No | Discharging intraoral wound |
| 12 | M | 25 | Mandible | Left body/angle/condyle | Yes | No | |
| 13 | M | 42 | Mandible | Right ramus/condyle | Yes | No | Discharging intraoral wound |
| 14 | F | 22 | Mandible | Right body/angle/condyle | Yes | No | Discharging intraoral wound |
| 15 | M | 17 | Mandible | Left body/angle/condyle | Yes | No | Multiple discharging extraoral fistulae |
| 16 | F | 21 | Mandible | Bilateral body | Yes | No | |
| 17 | F | 27 | Mandible | Bilateral body/condyle | Yes | No | Pain for 8 years; discharging intraoral wound |
| 18 | M | 19 | Mandible | Bilateral body, left angle/condyle | Yes | No | |
| 19 | F | 30 | Mandible | Right body | Yes | No | Discharging intraoral wound |
The median age of the patients was 24 years. The oldest was aged 70 years and the youngest was 17 years ( Fig. 1 ).
Seven patients reported pain elsewhere in the body. These areas included the neck, back, and ‘body’. One patient reported bilateral hip pain in addition to back pain, and one patient reported chest pain while coughing. Eleven of the patients reported having fever and fatigue.
Nine patients showed lesions on the left side and four showed lesions bilaterally, but none had bilateral condylar lesions. Five patients had right-sided lesions. One patient had a lesion in the anterior body region. Thirteen of the patients had an actively draining extraoral fistula and one patient had a closed extraoral fistula. Thirteen of the patients had condylar lesions. Eight other patients had posterior body/angle/ascending ramus lesions.
Only one patient had an isolated anterior lesion, and further examination of his clinical photographs and history, with no description of a draining sinus or exposed bone, suggests that the destruction seen on CBCT was related to the severe periodontal condition around his lower incisors. His clinical details suggest that his lesion may represent a different pathology to that of the other patients in the group.
Distinctive swelling, actively discharging fistulae, and scarring in the angle region of the mandible were present in 14 of the 19 patients of which 4 were bilateral ( Figs 2A and B, 3A and 4A and B ). A characteristic ascending ramus split and medial condylar destruction was frequently seen in the 17 CBCTs that were available ( Figs 2C–E, 3B–D, 4C and D and 5A–C ) .
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