Tubercular Osteomyelitis of Maxilla

Meher R^[1], Garg A^[2]

1. Ravi Meher (corresponding author)
M.S., DNB (ENT)
Assistant Professor(ENT)

2. Ashu Garg
M.S., DNB (ENT)
Senior resident, MAMC and Lok Nayak Hospital, Delhi.

Address for Correspondence
Dr Ravi Meher
Assistant professor
Himalayan Institute of Medical Sciences, Jollygrant, Dehradun,
Uttranchcal
India
E mail- meherravi@hotmail.com


ABSTRACT
Although tuberculosis of long bones and vertebral column is not uncommon, it is rare in small flat bones of face. We describe a case of tubercular osteomyelitis of maxillary sinus and discuss the clinical features and its management.

Keywords : tuberculoisis, osteomyelitis, maxilla, sinus


INTRODUCTION
Tuberculosis (TB) is an infection caused by Koch's bacillus (Mycobacterium tuberculosis, humanis or bovis). Its most frequent localization is in the lungs. The rare localizations include the sinuses, nasopharynx, nose and facial bones. It is sometimes confused with granulomatous or neoplastic processes for which reason diagnostic suspicion is important. Although the incidence of mycobacterial diseases, especially the extrapulmonary type, is on the rise in many regions of the world[1], it still remains an underdiagnosed entity. This, in part, is because of the occasional uncommon clinical presentations with the involvement of atypical sites.

CASE RECORD
A 15 year-old male presented with non-healing sinus below the left eye for the past 3 years. It was associated with recurrent crusting and purulent, occasionally blood stained discharge. The patient gave history of a swelling initially around 2 months back, spontaneously burst. It was followed by the development of a non-healing ulcer, which had gradually increased in size. There was no history suggestive of any systemic illness in the patient. Physical examination revealed areas of ulceration involving the left infraorbital region. It was covered with necrotic slough and crusts (Fig 1). On cleaning the slough and the crusts, granulation tissue could be visualized on the bed of the ulcer. There was no cervical lymphadenopathy. The routine hematological and biochemical analyses were within normal limits. His ESR was 28 mm. in the first hour and ELISA for HIV was non-reactive. His chest X-ray was normal. Mantoux(Tuberculin test) at 48 hours using purified protein derivative was positive (26 mm x 24 mm). An incisional biopsy from the edge of the ulcer was performed, which revealed non-specific chronic inflammation. CT scan of PNS revealed destruction of the anterior wall of maxilla with soft tissue suggestive of granulation tissue in left maxillary sinus(fig 2). A biopsy from left maxillary sinus granulations using Caldwell Luc's approach showed the epitheloid cell granulomas with caseation suggestive of tuberculosis. Stain for AFB was negative. Culture of tissue revealed no growth. ELISA for antimicrobial-IgM was done to confirm the diagnosis and was found to be reactive in 1:100 dilutions. The initial course of injectable penicillin, gentamicin and metrogyl was changed shifted to anti-tubercular drugs. Four-drug therapy included isoniazid (300mg), rifampicin (450mg), pyrazinamide (1500mg) and ethambutol (1200mg) for 2 months, followed by isoniazid (300mg) and rifampicin (450mg) for 4 months. Patient responded well to the treatment and is currently in follow up.
 

DISCUSSION
Most cases of tubercular involvement of maxillary sinus are secondary to pulmonary tuberculosis. Athough direct spread of infection from neighboring structures like orbit, other paranasal sinuses, face and nasal mucosa has also been implicated. From lung, infection spreads either by hematogenous route or lymphatics. The facial bones are unusual site for it and maxillary tuberculer osteomyelitis is even rare. Only few cases of maxillary tuberculosis have been reported[2,3]. Patient in the above case gave no history of pulmonary tuberculosis, which was confirmed by a chest skiagram. Thus, this being an unusual presentation, tuberculosis was not kept as a probable diagnosis when the patient first presented to us and was put on a course of parenteral antibiotics. It was only on the basis of a histopathology report of the lesion (caseating granulomatous lesion) that the lesion was suspected to be tubercular. But, since the stain for AFB was negative, ELISA confirmed the diagnosis for antitubercular-IgM. A positive family history, patients residing in high prevalence area and infections not responding to conventional antibiotics are indications to suspect and investigate for tuberculosis. A raised ESR, highly positive mantoux and cytology usually clinch the diagnosis. Serological tests for the diagnosis of tuberculosis that are based on the recognition of serum IgM antibodies to selected mycobacterium antigens are useful in diagnosis of extra pulmonary tuberculosis[4]. These tests use ELISA techniques and offer similar sensitivity to that of sputum microscopy. Gene amplification by the polymerase chain reaction (PCR) has also been used with great sensitivity and specificity to identify mycobacterium DNA[4].
Clinically the tubercular osteomeylitis is characterized by lack of early symptoms. Aappearance of fluctuant swelling i.e. Pott's Puffy tumor is usually the first symptom[5]. In our case also the initially there was a fluctuant swelling in the infraorbital region which later on bursted producing discharging sinus as presenting complaint. Skin attachment, discoloration and sinus formation are late features[6].
The commonest radiological lesion of tubercular osteitis is a lytic lesion It may present as expanding, destructive lytic lesion with ill defined, irregular margins having at first a sclerotic border and later osteoporotic edge[7]. A "button sequestrum" or "bone sand" may be seen within the lesion[8]. A similar lytic lesion was seen in the CT scan of present case.

Treatment is mainly conservative with antitubercular drugs. Surgery is indicated in cases of extensive destruction, presence of secondary infection and intracranial involvement. Surgery is not indicated for small lesions and presence of sequestrum is also not a definite indication for surgery. During treatment, radiological evidence of repair lags behind clinical evidence of improvement. Prognosis depends on gravity of associated tubercular lesions and extent of local disease, but now a day with chemotherapy prognosis is usually good.

Conclusion
It can be concluded that tubercular osteomyelitis of maxilla is a rare clinical entity and otolaryngologist should always consider it in the differential diagnosis of patients with swelling or sinus in relation to maxilla. Diagnosis is by a high index of clinical suspicion more so in an endemic area, positive Mantoux test, radiological features, aspiration of swelling for bacteriological examination and biopsy for histopathological examination. Also newer techniques i.e. ELISA and PCR can help in establishing the diagnosis of extrapumonary tuberculosis.

References
1. Al-Serhani AM. Mycobacterial infections of the head and neck: Presentation and diagnosis. Laryngoscope2001; 111: 2012-2016.
2. Mahindra S, Bais AS, Sohail MA, Maheshwari HB. Granulomatous osteomyelitis of the maxillary sinus. Otolaryngol. 1979; 8:255-8.
3. Rosenquist JB, Beskow R. Tuberculosis of the maxilla: report of case. J Oral Surg. 1977; 35:309-10.

4. Daniel M T. Tuberculois. Harrison's Principles of Internal Medicine. Vol 1, McGraw Hill Inc. 13th edition p 714.
5. Meher R, Singh I, Raj A. Tuberculosis of zygoma. Int J Pediatr Otorhinolaryngol. 2003:67(12): 1383-1385.

6. Sachdeva OP, Gulati SP, Kakker V, Arora B. Tuberculous osteomyelitis of zygoma. Trop Doct. 1993, 23: 190-1.
7. Malhotra R. Tubercular Osteitis of Skull. Indian Pediatrics. 1993, 30: 1119-1123.
8. Ganguly PK. Radiology of Bone and Joint Tuberculosis with special reference to tropical countries. London, Asia Publishing house, 1963, p 62.

Legends

Figure 1 Clinical photograph showing sinus in infraorbital region.
 

Figure 2 Axial CT scan