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Case Report

Tubercular osteomyelitis of the mandible in a youngfemale

http://dx.doi.org/10.1016/j.ijmyco.2014.02.0022212-5531/� 2014 Asian-African Society for Mycobacteriology. Published by Elsevier Ltd. All rights reserved.

* Corresponding author. Address: Department of Internal and Pulmonary Medicine, SheriKashmir Institute of Medical SciencSrinagar 190011, J&K, India. Tel.: +91 194 2401353x2256; cell: +91 9419004822; fax: +91 194 2403470.

E-mail addresses: parvaizk@gmail.com, parvaizk@rediffmail.com (P.A. Koul).URL: http://www.skims.ac.in (P.A. Koul).

Please cite this article in press as: PA Koul et al. Tubercular osteomyelitis of the mandible in a young female. Int. J. Mycobacteriol. (20dx.doi.org/10.1016/j.ijmyco.2014.02.002

Parvaiz A. Koul *, Umar Hafiz Khan, Rafi Ahmad Jan, Tajamul H. Shah,Farhana Bagdadi, Sanaullah Shah

Department of Internal and Pulmonary Medicine, SheriKashmir Institute of Medical Sciences, Srinagar, India

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

Article history:

Received 10 February 2014

Accepted 20 February 2014

Available online xxxx

Keywords:

Tuberculosis

Mandible

Osteomyelitis

A 16-year-old female presented with a 6-month history of a gradually increasing swelling of

the left side of her face. A panoramic radiographic view of the mandible showed diffuse

radiolucency in the ramus of the mandible with a loss of cortication on the superior and

anterior portion of the condyle. The computed tomography (CT) scan revealed destruction

of the mandibular bone and a large retromandibular and inferior temporal fossa mass with

areas of breakdown. The biopsy was consistent with tubercular osteomyelitis. Antitubercu-

lar therapy resulted in a marked reduction of the size of the swelling over a 9-month period.

� 2014 Asian-African Society for Mycobacteriology. Published by Elsevier Ltd. All rights

reserved.

Introduction

Osteoarticular tuberculosis accounts for 1–2% of all the types of

bone tuberculosis. Bone tuberculosis forms about 10% of extra-

pulmonary tuberculosis, of which 50% occur in the spine [1].

Tuberculosis of the flat bones of the skull is uncommon and that

of the mandible is especially rare as it contains less cancellous

bone [2]. Most of the cases which occur are due to a tubercular

focus elsewhere in the body, but primary tuberculosis of the

mandible is a very rare phenomenon [3–6]. The diagnosis as

such is often overlookeddespite a high prevalence of the disease

in high-burden countries like India. This study reports a case of

primary tuberculosis of the mandible in a 16-year-old female.

Case report

A 16-year-old female patient was referred to the department

of Internal Medicine, SKIMS, a 750-bedded tertiary care

hospital, for evaluation of a left-sided preauricular facial

swelling. The swelling had started insidiously 6 months ear-

lier with a painless increase in its size. In addition, the patient

also complained of trismus. The patient denied any history of

tooth extraction or any oral trauma, and there was no per-

sonal or family history of any chronic ailment. Clinical exam-

ination revealed a thinly built young female with a 6 · 7 cm

sized swelling on the left side of her face in the preauricular

region that was firm to hard in consistency, non-fluctuant,

mildly tender with normal overlying skin. Due to the presence

of marked trismus, only restricted examination of the oral

cavity could be achieved which showed erythematous buccal

mucosa in the region of the molars on the left side with ten-

derness over the retro-molar region. The rest of the general

physical and systemic examination was normal. Her investi-

gation revealed hemoglobin of 9.6 g/dL with a TLC of 6.76/

cu. mm. with a differential count of 70% polymorphs, 23%

lymphocytes, 4% monocytes and 3% eosinophils. The ESR

es, Soura,

14), http://

Fig. 2 – CT showing rarefaction and destruction of bone with

a 6.3 · 4.5 mass in retromandibular and inferior temporal

fossa. The mass shows areas of breakdown and bony

fragments.

Fig. 3 – Histopathological examination of trucut biopsy

showing chronic inflammatory cells with granulation tissue

admixed with dead bone and focal epithelioid cell

granulomas and Langerhans giant cells.

2 I n t e r n a t i o n a l J o u r n a l o f M y c o b a c t e r i o l o g y x x x ( 2 0 1 4 ) x x x – x x x

was 45 mm in the first hour. Her routine kidney and liver

functions were normal. LDH was 320 U/L with serum calcium

of 9.5 mg/dL. A skiagram of the chest was normal. Gram’s

smear and Ziehl–Neelsen staining of the sputum was nega-

tive for any organisms. A 5-TU tuberculin skin test was posi-

tive with an induration of 20 mm after 48 h. An ultrasound

examination of the abdomen and pelvis was normal. A pano-

ramic view radiograph of the mandible (Fig. 1) showed diffuse

radiolucency in the ramus of the mandible with a loss of cor-

tication on the superior and anterior portion of the condyle.

Computerized tomography of the mandible revealed pro-

nounced rarefaction and destruction of bone with a large

mass measuring 6.3 · 4.5 in retromandibular and inferior

temporal fossa with areas of breakdown and bony fragments

(Fig. 2). A trucut biopsy was undertaken which showed

chronic inflammatory cells with granulation tissue admixed

with dead bone and focal epithelioid cell granulomas and

Langerhans giant cells, consistent with tubercular osteomy-

elitis (Fig. 3). She was put on antitubercular therapy for

9 months and the swelling exhibited a marked reduction in

size.

Discussion

This patient had features of tuberculosis and emphasizes its

consideration in the differential diagnosis of mandibular

swelling. The rarity of the condition is such that the condition

is limited to only a few case reports in the English literature.

Tubercular infection of the oral tissues can be primary or

secondary. Primary lesions develop when tuberculosis bacilli

are directly inoculated into the oral tissues of a person who

has not acquired immunity to the disease. These frequently

involve gingiva, tooth extraction sockets and buccal folds.

Secondary infection of oral tissues can result from either

hematogenous or lymphatic spread or from autoinoculation

by infected sputum and direct extensions from neighboring

structures. Most of the reported cases of mandibular tubercu-

losis are secondary to focus elsewhere in the body and pri-

mary tuberculosis of the mandible is a rare occurrence [3–5].

The rarity of mandibular tuberculosis has been attributed to

the paucity of cancellous bone in the mandible with the angle

and the alveolar regions being affected most frequently [6].

Fig. 1 – Panoramic radiograph of the mandible showing

diffuse radiolucency in the ramus of the mandible with loss

of cortication on the superior and anterior portion of the

condyle.

Please cite this article in press as: PA Koul et al. Tubercular osteomyelitisdx.doi.org/10.1016/j.ijmyco.2014.02.002

The infected sputum or in some cases infected milk serves

as a direct source of infection or the tubercular bacilli gain ac-

cess through a break in the oral mucosa which can be either

in the form of opened tooth socket because of extraction or

a mucosal abrasions or gingival margin or perforation of an

erupting tooth [7]. Other routes for the occurrence of infection

can be by extension from a nearby soft tissue lesion which in-

volves the underlying bone. Hematogenous seeding has also

been suggested [8]. In this case, there was no history of dental

extraction or any trauma to the oral cavity, and possibly the

site of entry might have been gingivitis as the patient’s dental

hygiene was not maintained.

Mandibular tuberculosis is often insidious [7] and patients

usually cannot recall when the symptoms started. In a few

cases, it appears as an acute inflammatory swelling which

fails to resolve by the use of conventional antibiotics. This pa-

tient had the swelling for more than 6 months before medical

of the mandible in a young female. Int. J. Mycobacteriol. (2014), http://

I n t e r n a t i o n a l J o u r n a l o f M y c o b a c t e r i o l o g y x x x ( 2 0 1 4 ) x x x – x x x 3

attention was sought. Since mandibular tuberculosis is rare,

clinicians frequently confuse this with a pyogenic abscess

and if a discharging sinus is present, it can be misdiagnosed

as actinomycosis.

Radiologically mandibular tuberculosis begins as an area

of rarefaction with trabecular blurring. Gradually erosion of

cortical bone occurs which is then replaced by soft granula-

tion tissue and subsequently a sub-periosteal abscess forma-

tion takes place culminating into a visible painful swelling.

The granulation tissue undergoes caseation necrosis leading

to liquefaction which may burst either intra-orally or outside

leading to multiple discharging sinuses mostly along the infe-

rior border of the mandible or sometimes in the pre-auricular

region [9]. Pathological fractures of the mandible or seques-

tration have also been reported [9]. Cavities and pathological

fractures can be evident on CT scanning [7]. Treatment of

mandibular tuberculosis is with antitubercular therapy of

four conventional drugs in the form of rifampicin, isoniazid,

pyrazinamide and ethambutol initially as an intensive regi-

men followed by rifampicin and isoniazid for a period of

9–12 months; however, WHO recommends a short course

therapy of 6 months because of the pauci-bacillary nature of

the disease [10].

This case emphasizes the consideration of tuberculosis in

the differential diagnosis of a mandibular swelling and osteo-

myelitis of the jaw.

Conflict of interest

None declared.

Please cite this article in press as: PA Koul et al. Tubercular osteomyelitisdx.doi.org/10.1016/j.ijmyco.2014.02.002

R E F E R E N C E S

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[2] S.A. Sachs, L. Eisenbud, Tuberculous osteomyelitis ofmandible, Oral Surg. Oral Med. Oral Pathol. 44 (1977) 425–429.

[3] J. Fukuda, Y. Shingo, H. Miyako, Primary tuberculousosteomyelitis of the mandible: a case report, Oral Surg. OralMed. Oral Pathol. 73 (1992) 278.

[4] G.T. Richard, F.B. Donald, Tuberculosis osteomyelitis ofmandible, Oral Surg. Oral Med. Oral Pathol. 18 (1964) 7–13.

[5] Masaru Imamura, Toshio Kakihara, Kohsuke Yamamoto,Chihaya Imai, Atsushi Tanaka, Makoto Uchiyama, Primarytuberculous osteomyelitis of the mandible, Pediatr Int. 46(2004) 736–737.

[6] S.A. Sachs, L. Eisenbud, Tuberculous osteomyelitis ofmandible, Oral Surg. 44 (1977) 425–426.

[7] A.D. Dinkar, V. Prabhdesai, Primary tuberculosis of themandible, Dentomaxillofacial. Radiol. 37 (2008) 415–420.

[8] N. Worsaae, J. Reibel, Tuberculous osteomyelitis ofmandibula, Br. J. Oral Maxillofac. Surg. 22 (1984) 93–98.

[9] K.B. Gupta, M. Manchanda, S.P.S. Yadav, A. Mittal, Tubercularosteomyelitis of mandible, Indian J. Tuberc. 52 (2005) 147–150.

[10] American Thoracic Society, CDC, Infectious Disease Societyof America. Treatment of tuberculosis, Morbidity andMortality Report: Recommendations and Reports, 2003;52(RR-11):1–77.

of the mandible in a young female. Int. J. Mycobacteriol. (2014), http://