Keywords
mandible - osteomyelitis - tuberculosis
Introduction
Primary tuberculosis (TB) of the mandible is rare. It represents less than 2% of the
skeletal locations.[1]
[2]
[3] TB of the mandible usually presents as a part of multifocal lesions in the body,
involving other bones and lungs.[4] A total of 1.6 million people died from TB in 2021 (including 187,000 people with
HIV). In 2021, an estimated 10.6 million people were infected with TB worldwide.[5] Mandibular TB is uncommon and often diagnosed late because of nonspecific clinical
presentation and the absence of pathognomonic signs.[6]
[7] The mainstay of treatment is antitubercular multidrug therapy. Surgery is indicated
in only some cases.[8]
[9] We report the case of a 14-year-old male patient managed for mandibular swelling,
with provisional diagnosis of benign mandibular etiology, likely ameloblastoma. Primary
mandibular TB was detected incidentally on the histopathological examination.[10]
Case
A 14-year-old male patient presented to us with a history of painless, gradually increasing
swelling on the right side of chin for 1 year ([Fig. 1A–D]). He did not have any history of trauma, discharge, sinus, or redness in the region
or any sudden increase in size. There was a scar of previous biopsies over the right
side of his chin. There was no history of TB or family history of any chronic disease.
Clinical examination revealed a swelling involving right parasymphysis and body of
mandible, measuring approximately 6 cm in length and 2 cm in width, extending from
right lateral incisor till the first molar, with normal overlying skin. On palpation,
the swelling was firm in consistency. It was noncompressible, nonfluctuant, and slightly
tender. The oral examination of the patient showed good mouth opening with normal
occlusion and fair hygiene. His canine and first premolar over the involved bone were
loose. There were no palpable lymph nodes. Systemic examination was normal.
Fig. 1 Pre- and postoperative pictures of facial profile of patient. (A) Preoperative frontal view. (B) Preoperative occlusion. (C) Preoperative right lateral view. (D) Preoperative left lateral view. (E) Postoperative frontal view. (F) Postoperative occlusion. (G) Postoperative right lateral view. (H) Postoperative left lateral view.
The blood investigations and serology were within normal limits. The orthopantomogram
([Fig. 2A]) showed a multilocular osteolytic lesion involving the right parasymphysis and body
of the mandible. Computed tomography (CT) scan ([Fig. 2B–F]) revealed a large multiloculated osteolytic expansive lesion arising from the right
parasymphysis and body of mandible measuring 52 × 20 × 18 mm. The posterior cortex
was not involved by the disease process. No enlarged lymph nodes were identified.
The patient had undergone biopsies of the lesion at two different centers on separate
occasions. These biopsies did not show any cellular atypia. However, they were inconclusive
for any definitive ethology. Chest X-ray was found to be normal. Positron emission
tomography scan was done—lytic lesion is noted in body of right mandible with soft
tissue and fat stranding—there is significant cortical breech and minimal periosteal
reaction known as osteomyelitis involvement. Few tiny, right-sided, cervical level
IB, Il and Bilateral level V nodes are noted and appeared reactive. Enlarged prevascular
lymph node is noted measuring 2.0 × 1.5 cm; similar left-sided hilar node is also
noted. Rest of the bones were visualized and bilateral lungs appeared unremarkable.
Remainder of the survey showed unremarkable tracer distribution. Our provisional diagnosis
was benign mandibular ethology, likely ameloblastoma.
Fig. 2 Radiological investigations. (A) Preoperative orthopantomogram showing the multilocular osteolytic lesion of right
parasymphysis and body of mandible. (B) Cone beam CT showing the lytic lesion involving right side of the mandible from
lateral incisor till first molar. (C) CT three-dimensional reconstruction showing multiloculated osteolytic expansive
lesion arising from the right parasymphysis and body of mandible. (D) CT plate showing involvement of anterior cortex of mandible. (E) CT plate showing sparing of posterior cortex of mandible. (F) CT plate showing involvement of right side of the mandible from lateral incisor
till first molar. CT, computed tomography.
Informed consent was taken from the patient and his father for publication. The surgical
management involved excision of the lesion with external approach ([Fig. 3A]). The specimen was greyish, gritty, and with destruction of the anterior cortex
of the mandible. The canine and first premolar were involved. The specimen measurements
were 5 × 2 × 1.8 cm ([Fig. 3B]). Corticocancellous iliac crest bone graft, measuring 5.5 × 2 cm was used for reconstruction
of the defect ([Fig. 3C]). A single, continuous, 2.5-mm miniplate was used for fixation of bone graft using
2.5-mm screws ([Fig. 3D]). The postoperative period was uneventful.
Fig. 3 Intraoperative pictures. (A) External approach for the lesion. (B) Surgically resected specimen measuring 5 × 2 × 1.8 cm. (C) Anterior surface of corticocancellous iliac crest bone graft measuring 5.5 × 2 cm.
(D) Single 2.5-mm continuous miniplate in situ for fixation of bone graft.
The histopathological analysis of the excised specimen revealed a granulomatous lesion,
consistent with tuberculous osteomyelitis. On gross examination, a 1.6 × 1.2-cm cystic
cavity was seen at one extreme, lined by necrotic material. On microscopy, multiple
sections showed epithelioid granulomas, giant cells, and caseous necrosis. Necrotic
bone was noted. Patient was started on category I of antituberculosis therapy (ATT)
as per The National Tuberculosis Elimination Programme regimen. It included intensive
phase therapy for 2 months, consisting of rifampicin, isoniazid, pyrazinamide, and
ethambutol, followed by rifampicin, isoniazid, and ethambutol as continuation phase
therapy for 4 months.
On follow-up ([Fig. 1E–H]) at 1 year after completion of ATT (18 months after surgery), the patient had no
complaints and no evidence of recurrence. He has good mouth opening, normal occlusion,
and good aesthetic outcome. Radiological evaluation ([Fig. 4]) showed integration of bone graft. Patient is now being worked up for dental rehabilitation.
Fig. 4 Postoperative OPG. Postoperative OPG showing integration of bone graft and miniplate
with screws in situ. OPG, orthopantomogram.
Discussion
Ameloblastoma is a benign odontogenic tumor.[11] It is usually asymptomatic but when it attains considerable size, it can present
with jaw expansion. Radiologically, it shows an osteolytic pattern. Approximately
80% of these tumors are found in the mandible, but the maxilla is infrequently affected.[12]
Oral TB may manifest as swelling, pain, loosening of teeth, displacement of tooth
buds, ulcers, granulomas, involvement of salivary glands and temporomandibular joint,
and tuberculous lymphadenitis.[13] The lesions of primary orofacial TB could be the only presentation of the disease.[14] Primary TB of the mandible is an uncommon entity which represents less than 2% of
all skeletal TB.[1]
[2]
[3] As the mandible contains less cancellous bone, the chances of involvement of the
mandible are very less in comparison to maxilla, except that the alveolar and angle
regions have greater affinity.[15] In orofacial region, the mandible is affected more often than the maxilla and the
angle and alveolus of the mandible are commonly affected areas.[7]
Different mechanisms of extension of the infection to the mandible involve direct
inoculation through dental extraction or lesions of mucosa or perforation of an erupting
tooth, extension from a nearby soft tissue lesion which involves the underlying bone
and hematogenous seeding.[16]
[17] Mandibular TB is often difficult to diagnose because of the uncommon and nonspecific
presentation and absence of pathognomonic signs. In a few cases, it appears as an
acute inflammatory swelling.[7] In countries like India, TB can never be left out of the differential diagnoses.
Elzouiti et al reported a patient of ameloblastoma whose mandibular TB was diagnosed
incidentally on histopathology, which is extremely rare.[6]
Radiologically, there is no characteristic appearance of TB.[16] Mandibular TB starts as an area of rarefaction and trabecular blurring. Slowly,
there is erosion of cortical bone and it is replaced by soft granulation tissue and
subsequently subperiosteal abscess formation takes place ending with a visible painful
swelling. Pathological fractures of the mandible or sequestration have also been reported.[18]
The cases with minimally destructive lesions of TB can be managed by ATT. The medical
treatment includes rifampicin, isoniazid, pyrazinamide, and ethambutol initially as
an intensive phase regimen followed by rifampicin, isoniazid, and ethambutol as continuation
phase for a total period of 6 to 18 months with clinical and biological monitoring.[19] Surgical modalities described are for abscess developing in the soft tissue and
removal of the sequestered necrotic bone. Decortication of bone is indicated for moderately
destructive lesions because of medullary bone destruction and/or cortical bone perforation.[2] Early detection of the disease results in complete cure and can lead to reversal
of all destructive bony changes. If detected late, this can lead to serious complications
like tuberculous meningitis.[4]
Difficulty in differentiating primary tuberculous osteomyelitis of mandible from ameloblastoma
stems from the fact that both can present as slowly growing expansile osteolytic lesion
in the mandible. Discharging sinuses and signs of TB elsewhere in body may point to
tuberculous osteomyelitis. But as in our case, primary tuberculous osteomyelitis of
mandible may present without any of these. Bone sequestration, abscess formation are
common in tuberculous involvement, however these are not pathognomic and can rarely
be found in ameloblastoma. Definitive diagnosis in absence of systemic findings of
TB rests on histopathological examination.
We have carried out a review of 20 cases of primary TB of the mandible ([Fig. 5], [Table 1]) reported in literature.[3]
[6]
[14]
[15]
[16]
[20]
[21]
[22]
[23]
[24]
[25]
[26]
[27]
[28]
[29]
[30]
[31]
[32]
[33]
[34] We excluded all the cases which had signs of secondary involvement of the mandible
and included cases which had no signs suggestive of TB anywhere else in the body.
Although tuberculous osteomyelitis is more prevalent in endemic areas, it is present
all over the world and among all age groups. It usually begins as a localized swelling
and is associated with pain and enlarged cervical lymph nodes. While cytological examination
is mostly inconclusive, biopsy could provide the necessary information to diagnose
tuberculous osteomyelitis. However, biopsy is not always conclusive for TB, like in
our scenario. Radiological imaging with findings like radiolucency, bone destruction,
osteolytic and expansile bone lesions or sequestrum should raise the suspicion of
osteomyelitis. In cases with raised erythrocyte sedimentation rate and positive tuberculin
test, along with findings of osteomyelitis on imaging and pathological examination,
one should always rule out TB in endemic regions. We observed that ATT was sufficient
for complete resolution of the symptoms in most of the studies. Surgery was done in
cases where there was diagnostic uncertainty.
Table 1
Analysis of reviewed case reports and studies
|
Publication (author/year)
|
Patient demography age/sex
|
Main presenting symptom Swelling (Location/Tender/Duration)
|
Enlarged neck lymph nodes
|
FNAC (conclusive y/n)/na
|
Biopsy (specific/nonspecific granuloma)
|
OPG/CT/MRI (radiolucent-osteolytic/expansile/bone destruction/sequestrum)
|
Other investigations/Evidence before surgery in pointing towards TB
|
Management
|
Final HPE
|
Follow-up/recurrence
|
Comment
|
|
Nwoku et al, 1983, Nigeria[20]
|
8-year-old female
|
LJ/y/4 years
|
y
|
na
|
y/n
|
y/na/y/na
|
–
|
ATT
|
–
|
Complete resolution
|
–
|
|
Sepheriadou-Mavropoulou and Yannoulopoulos 1986, Greece[21]
|
35-year-old male
|
RJ/y/na
|
na
|
na
|
na
|
y/na/na/na
|
Chest X-ray, routine lab parameters normal
|
Curetting, removal of sequestra, ATT after HPE diagnosis
|
F/s/o Tuberculous osteomyelitis
|
Complete resolution, no recurrence
|
–
|
|
Fukuda et al, 1992, Japan[22]
|
76-year-old female
|
LJ/y/1 week
|
y
|
na
|
na
|
y/na/y/na
|
ESR = 80 mm/hr, tuberculin test positive
|
Saucerization done
ATT given
|
Destruction of bone, marrow fibrosis. Connective tissue with Langerhans giant cells,
epithelioid cells
|
No recurrence
|
–
|
|
Soman and Davies, 2003, United Kingdom[23]
|
37-year-old female
|
LPA/na/na
|
na
|
na
|
na
|
n/na/na/na
|
Sputum neg, ESR = 37 mm/hr,
Montoux positive,
Heaf test strong grade 3 reaction
|
TMJ exploration with shave of condyle
ATT after HPE
|
F/s/o tuberculous
|
Resolution
|
–
|
|
Modali et al, 2003, India[24]
|
19-year-old female
|
LJ/y/na
|
na
|
na
|
na
|
y/na/na/na
|
ESR = 50 mm/hr, AFB neg, Tuberculin test neg
|
Patient underwent curettage first, then hemimandibulectomy at 1 year
ATT was started after the second procedure
|
F/s/o tuberculous osteomyelitis
|
Recurrence after first procedure
Resolution after second procedure and starting of ATT
No recurrence after that
|
Patient was probably uncooperative and was not following up as advised
|
|
Dinkar and Prabhudessai, 2008, India[16]
|
10-year-old female
|
LJ/y/2 months
|
n
|
y
|
y/n
|
y/na/na/na
|
ESR = 22 mm/hr, tuberculin test neg
|
ATT
|
–
|
Complete resolution
|
–
|
|
Helbling et al, 2010, Switzerland[25]
|
22-year-old female
|
LPA/y
|
na
|
na
|
n/y
|
y/y/y/na
|
Sputum AFB neg, chest X-ray normal PCR positive
|
ATT
|
–
|
Complete resolution
|
–
|
|
Kumar et al, 2011, India[26]
|
9-year-old female
|
LJ/na/na
|
y
|
n
|
na
|
y/na/na/na
|
Montoux test positive (12 mm), chest X-ray and CT were normal
|
Surgical drainage with excision of coronoid process
ATT started after HPE
|
F/s/o tuberculous osteomyelitis
|
Complete resolution, no recurrence
|
–
|
|
Upadhyay et al, 2011, India[3]
|
14-year-old female
|
RPA/na/2 months
|
na
|
n
|
na
|
y/na/y/na
|
–
|
ATT
|
Epithelioid cell granulomas with central caseous necrosis with Langerhans giant cells
|
Complete resolution, no recurrence
|
–
|
|
Karjodkar et al, 2012, India[15]
|
45-year-old male
|
RJ/y/2 months
|
na
|
y
|
na
|
y/na/y/na
|
Montoux positive
PCR positive
|
ATT
|
–
|
Resolution and bone healing
|
–
|
|
Bai and Sun, 2014, China[27]
|
31-year-old male
|
B/L J/y/2 months
|
y
|
na
|
n/y
|
y/na/na/na
|
ESR = 28 mm/hr, tuberculin test 21 mm
PCR positive
|
Curettage of fistula and did PCR on yellowish white secretions
ATT given
|
–
|
Resolution
|
–
|
|
Koul et al, 2014, India[28]
|
16-year-old female
|
LPA/y/6 months
|
na
|
na
|
y/n
|
y/na/y/y
|
ESR = 45 mm/hr
|
ATT
|
–
|
Resolution
|
–
|
|
Gupta et al, 2016, India[29]
|
66-year-old male
|
LPA/y/1 year
|
y
|
na
|
y/n
|
y/na/y/na
|
Raised ESR, tuberculin test positive 24 mm in first 48 hrs
|
ATT
|
–
|
No recurrence
|
–
|
|
Sambyal et al, 2016, India[14]
|
3-year-old female
|
RJ/n/2 months
|
y
|
na
|
n/y
|
y/y/y/y
|
S-100, CD1A, PCR for TB neg
ESR = 47 mm/hr, tuberculin test positive
|
ATT
|
Osteomyelitis
|
Resolution of lesion, no recurrence
|
–
|
|
Dalmia et al, 2016, India[30]
|
21-year-old female
|
RPA/4 to 5 days
|
na
|
n
|
na
|
na/na/na/na
|
ESR normal, AFB neg TB PCR positive
|
ATT
|
Osteomyelitis of right hemi mandible with masseteric abscess
|
Complete resolution
|
–
|
|
Towdur et al, 2017, India[31]
|
49-year-old female
|
LPA/y
|
na
|
na
|
na
|
na/na/y/na
|
Blood ix, HIV, Montoux and chest X-ray neg
|
Condylectomy with curettage
|
F/s/o tuberculous
|
–
|
–
|
|
Kalaiarasi et al, 2018, India[32]
|
14-year-old male
|
RPA/na/na
|
y
|
n
|
y/n
|
y/na/na/na
|
ESR = 60 mm/hr, triple H negative, chest X-ray normal, AFB culture neg, sputum AFB
neg
|
ATT 6 months
|
–
|
Complete resolution
|
–
|
|
Elzouiti et al, 2021, Morocco[6]
|
50-year-old female
|
LJ/na/2 years
|
y
|
na
|
na
|
y/y/na/na
|
Chest X-ray normal
|
Left hemimandibulectomy
|
Ameloblastoma with mandibular and lymph node tuberculosis
|
Resolution of lesion, no recurrence
|
Simultaneous ameloblastoma with tuberculosis
|
|
Prashant et al, 2022, India[33]
|
14-year-old female
|
RPA/y/2 months
|
n
|
na
|
na
|
y/na/y/na
|
GeneExpert TB, culture positive
|
Sequestrectomy, curettage of necrotic tissue in right condylar region
|
TB
|
–
|
–
|
|
Gupta et al, 2022, India[34]
|
19-year-old female
|
RJ/n/2 months
|
na
|
n
|
na
|
y/na/na/y
|
Raised ESR, Montoux neg, chest CT normal, sputum AFB neg
|
Exploration, decortication
|
Suggestive of tuberculosis
|
Complete resolution
|
–
|
Abbreviations: AFB, acid fast bacilli; ATT, antituberculosis therapy; B/L J, bilateral
jaw; ESR, erythrocyte sedimentation rate; HPE, histopathological examination; hr,
hour; LJ, left jaw; LPA, left preauricular area; mm, millimeter; MRI, magnetic resonance
imaging; n, no; na, not mentioned in publication; neg, negative; PCR, polymerase chain
reaction; RJ, right jaw; RPA, right preauricular area; TB, tuberculosis; TMJ, temporomandibular
joint; y, yes; f/s/o, features suggestive of.
Conclusion
We have shared our experience of incidental detection of primary mandibular TB in
a patient presenting with ameloblastoma-like picture. Clinical and radiological presentation
of primary mandibular TB is not specific, making it a diagnostic challenge for clinicians.
The histopathologic analysis is diagnostic for tuberculous osteomyelitis. It should
form a part of differential diagnoses in endemic areas. If biopsy reveals tuberculous
osteomyelitis, ATT can avoid the need of surgical intervention.
Fig. 5 Comparative chart showing the number of patients with various findings in terms of
enlarged lymph nodes, radiological finding, FNAC, biopsy, evidence pointing towards
TB, gender and age of the patient. ESR, erythrocyte sedimentation rate; TB, tuberculosis;
yrs, years; FNAC, fine needle aspiration cytology.
