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Advances in Oral and Maxillofacial Surgery 2 (2021) 100064

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Advances in Oral and Maxillofacial Surgery


journal homepage: www.editorialmanager.com/adoms/default.aspx

Case report

Tuberculous osteomyelitis in condyle of mandible: A case report


Niranjala Mohad a, *, Ashok Dabir b, Jayesh Vahanwala c, Mukul Padhye d, Jayant Patwardhan a
a
Oral and Maxillofacial Surgeon, Mumbai, India
b
Oral and Maxillofacial Surgeon, Panel Consultant- Breach Candy Hospital, Shushrusha Hospital, Mumbai, India. Emeritus Professor, Dr. D.Y. Patil Dental College &
Hospital, Navi Mumbai, India
c
Dept. of Oral and Maxillofacial Surgery, Vaidik Dental College and Research Centre, Daman, India
d
Dept. of Oral and Maxillofacial Surgery, Dr. D.Y. Patil Dental College & Hospital, Navi Mumbai, India

A R T I C L E I N F O A B S T R A C T

Keywords: Tuberculosis (TB) is a chronic infectious granulomatous disease caused by the air-borne bacillus Mycobacterium
Tuberculous tuberculosis (M.tuberculosis, M.Tb), and less frequently by other bacterium in the M. tuberculosis complex (M.
Osteomyelitis bovis, M.africanum).
Mandible
It has two forms pulmonary and secondary causing other kinds of TB, collectively denoted extra pulmonary
Condyle
tuberculosis (EPTB), constitutes 15% to 20% of all cases of TB among immunocompetent adults, and it accounts
Infectious osteomyelitis
for more than 50% of the cases in human immunodeficiency virus (HIV) positive individuals.
Maxillofacial manifestations of tuberculosis form nearly 10% of all extra pulmonary manifestations of the
disease. Tubercular infection of mandible is a rare condition. Tuberculous involvement of mandibular condyle is
even rarer. The clinical appearance of TB infection of the TMJ has been described as unspecific, resembling
arthritis, osteomyelitis, cancer or any kind of chronic joint diseases. The only manifestation may be a localized
painful swelling of the jaw. The presented case is of osteomyelitis of the mandibular condyle in a 12year old male
patient for whom TB was later suspected.
In this case report the importance of correct diagnosis is emphasized as Osteomyelitis of condyle has the risk of
being easily missed, owing to its atypical signs, symptoms and radiographic appearance.

Introduction incidence of the disease mainly because of the emergence of multidrug-


resistant strains of Mycobacterium tuberculosis (MDR-TB) and its associa-
Tuberculosis (TB) is one of the oldest diseases known to affect tion with HIV infection, leading to the spread of this disease in immu-
humans [1]. nocompromised and AIDS-positive individuals [6].
It is a chronic infectious granulomatous disease caused by air-borne Susceptibility to TB in developing countries results from multiple
bacillus Mycobacterium tuberculosis (M.tuberculosis, M.Tb), and less variables, including poverty, economic recession, malnutrition, and
frequently by other bacterium in the M. tuberculosis complex (M. bovis, multidrug resistance. Although, it does not make exception of rich and
M. africanum) [2,3]. On March 24, 1882, Dr. Robert Koch announced the well nourished people too.
discovery of Mycobacterium tuberculosis, the bacterium causing TB. It is It is very important to diagnose this disease at an early stage. If left
characterized by fever, weight loss, Palmer and night sweat. untreated, it can prove fatal within 5 years in more than half the cases [7,
Tuberculosis (TB) continue to be India's severest health crisis as it kills 8]. Early detection and prompt treatment are crucial for management of
an estimated 480,000 people every year [4]. To achieve the targets and this highly infectious and communicable disease.
milestones set by the World Health Organization to their ‘End TB Strat- The lung is the predominant site of TB. Primary pulmonary TB should
egy’, to stop the global TB epidemic by 2035 and India's commitment to be distinguished from post primary pulmonary TB, which is the most
eliminate this disease from the country by 2025 [5], it will be important frequent manifestation of TB in adults (70%–80% of cases) [9]. Pulmo-
to improve the case finding and effectively treat cases of tuberculosis nary tuberculosis is often the primary lesion. Tuberculous infection can
both in the public and private sector. spread through local inoculation, lymphatics, or hematogenous route, if
Furthermore, in recent years there has been an upsurge in the the patient's immune response is poor.

* Corresponding author. Oral and maxillofacial surgery, Mumbai, India.


E-mail addresses: drniranjalamohad28@gmail.com (N. Mohad), drjvahanwala@gmail.com (J. Vahanwala).

https://doi.org/10.1016/j.adoms.2021.100064
Received 28 February 2021; Accepted 5 March 2021
Available online 10 March 2021
2667-1476/© 2021 Published by Elsevier Ltd on behalf of British Association of Oral and Maxillofacial Surgeons. This is an open access article under the CC BY-NC-ND
license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
N. Mohad et al. Advances in Oral and Maxillofacial Surgery 2 (2021) 100064

Extrapulmonary tuberculosis (EPTB) is an uncommon form of chronic osteomyelitis of the Mandibular Condyle.
infection, which does not present with the typical signs and symptoms of
pulmonary tuberculosis. The host inflammatory reactions play an
Case report
important role in protection from this disease. In EPTB highly vascular
areas such as lymph nodes, pleura, genitourinary tract gastrointestinal
A 12 year old boy reported to us with complain of swelling on right
system, central nervous system, bones,joints, meninges, skin and oral
side of face in the preauricular region and difficulty in mouth opening. He
cavity are commonly affected. EPTB generally has an insidious presen-
had a history of swelling since 2 months which was slowly increasing in
tation, a slow evolution, and paucibacillary lesions and/or fluids. Access
size. He consulted a local dentist, who prescribed antibiotics and anal-
to the lesions through secretions and body fluids is not always possible;
gesics, but neither there was reduction in the size of the swelling, nor
therefore, invasive techniques may be necessary to obtain material for
symptomatic relief. He had no prior medical history of treatment for any
diagnostic investigation [10].
chronic infective disease. In family history both parents had a history of
Nassau et al. introduced the enzyme-linked immunosorbent assay
tuberculosis during their childhood and were treated for the same.
technique for the detection of antibodies against M. tuberculosis. Various
Patient was moderately built and well nourished. The patient pre-
strategies exploiting DNA, RNA, proteins, fluorescent staining of nucleic
sented with trismus and a diffuse swelling on right side of face in the
acid, and other biomolecule-based products have been developed for
preauricular region with no sinus or discharging pus. Overlying skin was
early-stage and rapid diagnosis of pulmonary and extrapulmonary TB.
normal in colour and ear lobule was not elevated. There was a localized
For detection of antibodies in the early stages of TB infected individuals,
rise in temperature. Swelling was tender and soft on palpation. Tender-
secretory protein antigens or culture filtrate protein (CFP) antigens of
ness was elicited in the temporalis muscle of the same side with no
M. tuberculosis can also be used [11].
Obvious lymphadenopathy of the head and neck. Presence of trismus
Oral TB may be either primary or secondary. In primary form, the oral
restricted mouth opening to 15mm. Examination of oral cavity was
cavity is the initial site of infection as a result of direct inoculation of
normal. (Fig. 1).
bacteria. The lesions of primary orofacial TB are uncommon, and
Provisional differential diagnosis of chronic suppurative osteomye-
generally arise in young individuals, presenting as a single painless ulcer
litis of the condyle was suspected.
with regional lymph node enlargement. It is reported that 60% of all
In radiographic findings panoramic view showed diffuse radiolucency
cases of TB of the jaw occur in children below the age of 16 years,
with loss of cortication on the superior and anterior portion of condyle,
whereas secondary orofacial TB arises subsequent to the spread of
the extent of destruction observed lead to the suspicion that a larger
tuberculous infection from another site through the lymphatic and he-
lesion may be present. Cone beam computed tomograph (CBCT) of the
matogenous routes [12].
Right TMJ showed pronounced rarefaction and destruction of bone in
Tubercular osteomyelitis of mandible is rare. The clinical presenta-
mandibular condyle with discontinuity of the cortical boundary sugges-
tion of TB infection of TMJ is considered as Master Mimic, thus can
tive of perforation and erosion of the condylar head (Fig. 2).
resemble arthritis, osteomyelitis, or any other kind of chronic joint dis-
Radiographic diagnosis came as osteomyelitic changes in relation to
ease. In absence of systemic infection, the diagnosis of primary infection
right condyle.
of TMJ is challenging. If the diagnosis is missed and the appropriate
Preoperative, Chest Physician referral and all routine investigations
treatment is delayed, osteoarthritic-like changes and the degree of joint
like CBC, HIV, HbsAg, HCV, RBS, HbA1c, LFT, RFT, Coagulation profile,
destruction can be severe [13].
Blood grouping, TSH, Chest Xray, and ECG were done.
We present a case of a 12year old boy with primary tuberculous
The patient was operated under General Anaesthesia with

Fig. 1. Preoperative clinical pictures.

2
N. Mohad et al. Advances in Oral and Maxillofacial Surgery 2 (2021) 100064

Fig. 2. Preoperative Scans.

Nasotracheal intubation. On Aspiraton, thick yellow pus and blood was curettage of the necrotic tissue performed in the right condylar region.
aspirated from the swelling and the smear showed abundant caseous The excised tissue was sent for histopathologic examination, which
necrosis, occasional epitheloid cell granulomas, aggregated polymorphs showed epithelioid cell granulomas, histiocytes and multinucleate
and few lymphocytes. These findings were strongly suggestive of Langerhans giant cells, with a central acidophilic necrotic focus and
Tuberculous etiology(Fig. 3B). surrounded by lymphocytes (Fig. 4). Histopathologic report confirmed
A preauricular incision was made along the natural crease anterior to the diagnosis of TB.
the tragus (Fig. 3)dissection continued along the cartilage of external Additional, Genexpert TB test and culture test were advised. Myco-
auditory canal in order to prevent damage to the Auriculotemporal nerve bacterium tuberculosis complex was detected in Genexpert TB and culture
and Superficial temporal artery. Superficial layer of temporalis fascia was test.
incised along the zygomatic arch and blunt dissection carried out to Final diagnosis of extra pulmonary tuberculosis with osteomyelitis of
expose the lesion. Through this approach sequestrectomy and thorough Right mandibular condyle was confirmed. Patient continues to see Chest

Fig. 3. Intraoperative surgical pictures.

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N. Mohad et al. Advances in Oral and Maxillofacial Surgery 2 (2021) 100064

Fig. 4. Postoperative clinical and Histopathology slide picture.

Physician for Anti Tubercular Treatment (ATT) regimen {Rifampicin kept on periodic follow up. On 3 months postoperative follow up, the
(600 mg) þ Isoniazid (300 mg), Ethambutol(800mg), Pyr extra oral swelling had resolved completely, mouth opening improved
azinamide(750mg), Vitamin B6, D, calcium supplements } and was from 15mm (pre-op) to 40mm (post-op), however mild deviation toward

Fig. 5. 3 Months Postoperative Clinical pictures.

4
N. Mohad et al. Advances in Oral and Maxillofacial Surgery 2 (2021) 100064

right side was seen (Figs. 4 and 5). Panoramic radiographs (Fig. 6) The diagnosis must be established by histologic examination of the
showed progressive bone healing in right condylar head. tissue and evidence of the organisms in the lesion, in sputum or tissue
smears. The site specific tissue or fluid aspirate must be submitted for
Discussion smear culture and histological examination as has been done in this case
[13].
Tuberculosis (TB) is a chronic, infectious granulomatous disease In the study done by Dileep et al., in 201,711, a method of exploring
caused by Mycobacterium tuberculosis and less frequently Mycobacterium CFP antibodies (polyclonal chemically conjugated with liposome parti-
bovis and atypical mycobacteria [14], infects 20–43% of the world's cles of 0.2–0.4 mm) that bind with corresponding antigenic determinants
population. It ranks among the top 10 causes of death world-wide [15]. available on clinically confirmed serum and tissue biopsy specimens,
India accounts for nearly one third of the global burden of TB [16]. The which results in visible agglutination reaction, has been developed.
average prevalence of all forms of TB in the country is estimated to be The developed diagnostic card is an effective diagnostic test for cases
5.05 per thousand and average incidence of smear-positive cases, 84 per that were found negative with smear/culture owing to a low number of
lakh [17]. Without greater control efforts, the annual global incidence of microorganisms or because patients were undergoing a drug therapy.
TB may increase by 40% by end of 2020 [18]. The fundamental principle of this newly developed diagnostic card is
Tubercle bacilli are mainly human and bovine. According to Western to evaluate the antigen concentration of active M. tuberculosis or the
reports, bovine tubercle bacilli are responsible for 80% of Osteoarticular breakdown product of M. tuberculosis present in specimens (serum or
lesions in Patients younger than age of 10 years. The human bacillus is tissue biopsy) [11].
responsible for almost all cases of osteoarticular TB in India. First the The relatively new rapid cartridge based, fully automated, nucleic
primary complex develop consisting of local infection and the infected acid amplification test (CB-NAAT)approved by WHO, which simulta-
regional lymph nodes. During this process, tubercle bacilli are carried to neously detects M. tuberculosis DNA as well as mutation that confer
other organs via the blood and lymphatic vessels. They may be the origin rifampicin resistance, with a high sensitivity and specificity, can provide
of both extrapulmonary TB and reactivation TB in some individuals [19]. results in less than 2 hours compared with the turn around time of 8–10
An oral tuberculous lesion may be primary or secondary [12]. Pri- weeks with conventional drug sensitivity testing, making it useful in
mary lesions occur when oral inoculation occurs first and there is no diagnosis of tuberculosis and in screening the cases for MDR-TB [23].
concurrent pulmonary involvement. Table 1 describes various diagnostic test for TB and their advantages.
Only few primary manifestation of TB in TMJ has been reported till
Primary inoculation can occure by date [13]. These Rare lesions can pose a diagnostic challenge; TB of the
TMJ is unlikely to be diagnosed early in the nondestructive stage, espe-
1. Sputum, cow raw milk, infected food or by a contaminated object cially when there are no signs, symptoms and radiographic evidence of
(eg,utensil), through an opening in the gingiva by an erupting tooth TB elsewhere in the body.
or an extraction socket or through an open pulp in a carious tooth About half of such patients do not have pulmonary disease at the time
2. Spread from an overlying soft tissue lesion [12]. they report, as the primary infection heals leaving some surviving tu-
bercle bacilli. With a lowering of resistance, these are reactivated pro-
Primary orofacial TB can affect both soft and hard tissue. ducing local as well as heamatogenous spread [7]. The condyles are more
Soft tissue presentation of oral tuberculosis in more than 40%of cases prone to tubercular involvement because of their cancellous portion.
is described as a typical ulceration, superficial or deep, with irregular Infection begins in the subchondral region and spreads to involve the
margins, undermined edges and a granulating floor. It tends to increase cartilage, synovium, and joint space [19].
in size slowly and, sometimes, may be ragged and indurated. The Andrade et al. studied 46 cases of Oro-facial tuberculosis over a
appearance of satellite lesions is rare. Infrequently, an oral TB lesion may period of 16 years and reported no gender predilection (M: F ¼ 0.917)
appear as a non-tender swelling, a nodular mass or diffuse granulomatous and a wide age range of occurrence, the most common age group being
enlargement. Although the clinical picture of oral TB is variable and 11–30 years.The 10 point protocol given by Andrade et al., dictates
nonspecific, the most common presentation is ulceration. Therefore, a guidelines for investigations, diagnosis, treatment and follow up for Oro-
complete medical history, clinical examination and accessory in- facial TB cases [24].
vestigations must be undertaken to differentiate a tuberculous ulcer from In our case report, it is clear that the patient had involvement of the
other diseases that present as ulcers during their course [20]. Temporomandibular joint early in the course of his clinical infection,
Morgagni reported the first case of oral TB seen as lingual involve- given the initial symptoms of pre auricular pain and the extent of
ment in 1761 21 condylar destruction on the radiograph. This would imply that the
Considering hard tissue presentation of Orofacial TB, there is rarity in infection may have been initiated in the condyle or joint space and
cases affecting the Mandible, Temperomandibular Joint and Hard palate. infected the surrounding hard and soft tissues secondarily.
In a rare case, the Bacillus Calmette-Guerin (BCG) vaccination has been Most of the time, swelling and pain are misdiagnosed as odontogenic
reported as a possible precursor for tubercular osteomyelitis of the hard infection and the patients are prescribed antibiotics. Ruggiero et al. [25]
palate [22]. stressed that benign and malignant neoplasms of the joint and infective
They are extremely rare such that published literature about primary processes such as acute suppurative arthritis, osteomyelitis, and chronic
lesions exists only in the form of case reports. Based on reviews published tuberculous arthritis have to be considered in such cases.
before 1950, oral TB accounted for 0.1–5% of all TB infections. With Indications for surgical treatment as an adjunct to chemotherapy
advances in chemotherapy and improvement in public health and hy- include unresponsiveness to and noncompliance with medical therapy
giene, the rate has decreased. However, nowadays, oral manifestations and the presence of a large or otherwise undrainable abscess [25].
are re-appearing alongside many forgotten extrapulmonary infections as The treatment of orofacial tuberculosis varies depending on its pre-
a consequence of the emergence of drug-resistant TB and the emergence sentation. In the literature, cases of minimal destructive lesions only
of Acquired Immuno-deficiency Syndrome (AIDS) [21]. required medical intervention of antitubercular therapy to achieve res-
In general, Primary TB lesion of jaws begins in the cancellous portion olution, precluding the need for surgical treatment of the lesion.
of the bone involved. TB involving the mandible is rare because it con- Moderately destructive lesions required decortication of bone as a result
tains less cancellous bone [19]. The diagnosis of TB of the mandible is of medullary bone destruction and/or cortical bone perforation.
very difficult because there are no specific pathognomonic signs of The chemotherapeutic regimen for extrapulmonary tuberculosis is
infection. The only manifestation might be a localized swelling of the the same as that for pulmonary tuberculosis The World Health Organi-
jaw, as seen in our patient, which could be misdiagnosed as an abscess. zation guidelines for appropriate TB chemotherapy include long-term

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N. Mohad et al. Advances in Oral and Maxillofacial Surgery 2 (2021) 100064

Fig. 6. Postoperative Scans.

published and due efforts will be made to conceal his identity, but ano-
Table 1
nymity cannot be guaranteed.
Diagnostic Tests for TB.
Sr.no DIAGNOSTIC TOOL ADVANTAGE
Funding
1 Heaf Test Easy interpretation; less inter-observer variability
2 Mantoux Test Screening test, diagnosis of active TB, more precise None.
than radiograph
3 Radiograph Easy to perform
4 Ziehl - Neelson Simple, non-invasive, economical
Declaration of competing interest
staining
5 Auramine More sensitive, quick results, contrast enables better
Fluorescence visualization None.
6 ELISA More sensitive, faster
7 LJ medium culture Less expensive than BACTEC
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8 BACTEC Differentiates M.tuberculosis from other species
9 PCR Sensitive, quick results
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