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Tuberculous osteomyelitis of zygoma: an unusual location
Vol. 116 No. 3 September 2013
Tuberculous osteomyelitis of zygoma: an unusual location Virendra Singh, MDS,a Shruti Khatana, MDS,b Pranav Gupta, MDS,b Amrish Bhagol, MDS,c and Anjali Narwal, MDS,d Haryana, India SHARMA UNIVERSITY OF HEALTH SCIENCES
Tubercular osteomyelitis of midfacial bones is extremely rare, although tuberculosis of long bones and the vertebral column is not uncommon. Because of the rare incidence, myriad presentation, and lack of specific symptoms, this condition presents a challenge in diagnosis and calls for acute clinical awareness. This article presents a case report of a 12-year-old girl with complaints of gradually increasing swelling lateral to and below her right eye for 4 months with pus discharge. She was treated with surgical curettage and 4-drug antitubercular therapy and responded with complete remission of the sinus. (Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:e156-e160)
Osteomyelitis of the craniofacial bones is an uncommon entity.1 The involvement of the bone in the middle third of the face is rare. The etiology most commonly arises from odontogenic sources or infected fracture sites and is usually pyogenic. Tubercular osteomyelitis of midfacial bones is extremely rare, although tuberculosis (TB) of long bones and vertebral column is not uncommon.2 Tubercular osteomyelitis is clinically and radiologically indistinguishable from pyogenic osteomyelitis and the 2 conditions can be differentiated only on the basis of histopathological evaluation of involved tissue.1 Because of the rare incidence, myriad presentation, and lack of specific symptoms, this condition presents a challenge in diagnosis and calls for acute clinical awareness. Representing a similar dilemma, we hereby report a case of tuberculous osteomyelitis of zygoma.
CASE REPORT A 12-year-old girl presented to the oral and maxillofacial surgery outpatient department of Government Dental College, Rohtak, India, with complaints of gradually increasing swelling lateral to and below her right eye for 4 months with pus discharge from the same for 15 days.
a
Professor and Head, Department of Oral and Maxillofacial Surgery, Government Dental College, Pt. B.D. Sharma University of Health Sciences, Haryana, India. b Resident, Department of Oral and Maxillofacial Surgery, Government Dental College, Pt. B.D. Sharma University of Health Sciences, Haryana, India. c Assistant Professor, Department of Oral and Maxillofacial Surgery, Government Dental College, Pt. B.D. Sharma University of Health Sciences, Haryana, India. d Assistant Professor, Department of Oral and Maxillofacial Pathology, Government Dental College, Pt. B.D. Sharma University of Health Sciences, Haryana, India. Received for publication Sep 20, 2011; returned for revision Dec 2, 2011; accepted for publication Dec 24, 2011. © 2013 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter doi:10.1016/j.oooo.2011.12.017
e156
A presumptive diagnosis of osteomyelitis was made and routine investigations and radiographs were ordered. Within a week of the presentation, the child complained of another fluctuant swelling on the right zygomatic prominence. Oral antibiotics from the penicillin group were started but no response was observed. Approximately 7 days later the child complained of redness of the right eye, which revealed conjunctival congestion, although the rest of the ophthalmologic examination was normal. On clinical examination, the swelling lateral and inferior to the right eye was firm and nontender on palpation with diffuse surrounding erythema. There was recurrent crusting and ulceration at the site, which on cleaning, revealed granulations at the margins and bed of the lesion. The overlying skin was adherent to the bone at certain areas. The other swelling that presented near the right zygomatic prominence measured 8 ⫻ 5 mm with fluctuation and discoloration that spontaneously burst and later persisted as a discharging sinus (Figure 1). The pus was sent for culture and sensitivity and did not show any growth/acid-fast bacilli (AFB)/sulfur granules. The patient had no systemic illness and was healthy otherwise. No history of trauma or previous infection was reported. There was no lymphadenopathy on examination. Routine blood investigations were normal. The erythrocyte sedimentation rate (ESR) was 50 mm/h in the first hour, hemoglobin was 12.0 g/dL, total leucocyte count (TLC) was 5500/mm3, differential leucocyte count (DLC) was P-58, L-36, M-6; enzyme-linked immunosorbent assay for HIV was negative; 48-hour Montoux reading was 27mm; and sputum for AFB was negative. On further inquiry, the patient’s father reported having been treated for pulmonary TB 1 year previously and was completely asymptomatic later. The child had no history of TB or any antitubercular therapy in past. The patient was Bacille Calmette-Guerin (BCG) vaccinated at infancy. Radiological investigations were done. Occipitomenton view revealed an ill-defined osteolytic lesion in the right zygomatic bone. Computed tomography (CT) scan of the face revealed few lytic lesions in relation to the right zygomatic bone with evidence of sclerosis and loculated subcutaneous soft tissue collection (Figure 2). Chest radiograph was normal.
OOOO Volume 116, Number 3
Fig. 1. Preoperative clinical photograph of the patient showing crusting and discharging sinus.
Fig. 2. CT scan of the face (coronal view) revealing few lytic lesions in relation to right zygomatic bone. The granulation tissue from the sinus was curetted intraorally under local anesthesia and the histopathology report revealed a caseating granulomatous lesion strongly suggestive of tuberculous osteomyelitis and was negative for AFB on special staining (Figure 3). The tissue specimen was also sent for culture, which was positive for mycobacterial growth by 5 to 6 weeks. The patient was put on a 4-drug antitubercular treatment (isoniazid, rifampicin, ethambutol, and pyrazinamide), and she responded with complete remission of the sinus. Her 6-month follow-up was uneventful (Figures 4 and 5).
DISCUSSION Tuberculosis is common in India and Southeast Asia, where the prevalence rate is about 4 in every 1000 people; 15% of the world’s tuberculous population resides in India.3
CASE REPORT Singh et al. e157
Fig. 3. Histopathological section showing chronic inflammatory infiltrate with giant cells (hematoxylin and eosin, original magnification ⫻10).
Fig. 4. Postoperative clinical photograph of the patient (6month follow-up).
Tuberculous osteomyelitis of the skull bones accounts for 0.1% to 3.7% of all cases of skeletal TB.4,5 Three-fourths of such cases were seen in patients younger than 20 years and half in those younger than 10 years.6 The present case was that of a 12-year-old girl. Most of the cases of TB of the skull are secondary to pulmonary TB7; however, there was no clinical or radiological evidence of pulmonary involvement in our case. Primary zygomatic TB is a rare condition and only a few cases have been reported.2 The presentation is nonspecific and usually confuses the diagnosis. A positive family history, endemic area, and infections not responding to conventional antibiotics should raise a suspicion. Raised ESR, highly positive Montoux, radiologic investigations, and histopa-
ORAL AND MAXILLOFACIAL SURGERY e158 Singh et al.
Fig. 5. Postoperative CT scan of the face (coronal view at 6-month follow-up).
thology are usually the key to diagnosis. A similar presentation was noted in our patient. Clinically, tuberculous osteomyelitis is characterized by lack of early symptoms.8 Appearance of a fluctuant swelling is usually the first symptom.8 Our case was characterized by 2 different appearances from the spectrum of presentations. The superior lesion was characterized by skin adhesion, erythema, crusting, and granulations, whereas the inferior lesion was typically fluctuant and spontaneously burst, producing a discharging sinus. The most common radiological presentation is a lytic lesion.8 The first demonstrable change in a small translucent area owing to decalcification is clinically undetectable at this stage. With the progress of the lesion, the decalcified areas increase in size with occurrence of bone destruction.2 It may present with a sclerotic border and later an osteoporotic edge.9 The dilemma is because radiologically the tuberculous osteomyelitis resembles nonspecific osteomyelitis.6 Our patient also presented with lytic lesions and sclerosis was visualized on the occipitomenton view and CT scan of the face. These findings can also be encountered in eosinophilic granuloma, congenital syphilis, or secondary malignant deposits.6 Such nonspecific presentation and overlapping symptoms make tuberculous osteomyelitis a diagnostic challenge, as in our case, where pyogenic osteomyelitis was the initial diagnosis. Moreover, extrapulmonary TB (EPTB) involves relatively inaccessible sites and because of the nature of the sites involved, fewer bacilli can cause much greater
OOOO September 2013
damage. The combination of small numbers of bacilli and inaccessible sites causes bacteriologic confirmation of a diagnosis to be more difficult, and invasive procedures are frequently required to establish a diagnosis.10 Diagnosis of EPTB is based on 1 culture-positive specimen from the extrapulmonary site, histologic evidence, or strong clinical evidence consistent with active EPTB disease followed by a medical officer’s decision to treat with a full course of anti-TB therapy.11 In our case, the histopathology report was highly suggestive of tuberculous osteomyelitis, although AFB was not demonstrated on special staining. The culture report was positive for mycobacterial growth by 5 to 6 weeks, although the chemotherapy had already been started based on histopathology and strong clinical suspicion. The good clinical response to chemotherapy obviated the need for any further diagnostics. Because of the relative scarcity of organisms within the tissue, the special stains successfully demonstrate the organism in only 27% to 60% cases.12 So, the absence of AFB in the tissue specimen does not rule out TB. The diagnostic pipeline for TB is replete with a multitude of tests but their relevance in terms of application and cost-effectiveness, especially in areas with a high burden of TB, such as India, Southeast Asia, and Africa, is yet to be established. Sputum smear microscopy, culture, biopsy and tubercular skin testing (TST) remain indispensable for tubercular diagnosis.13 A positive tuberculin test is only 1 piece of evidence in favor of the diagnosis of TB. The younger the child and the greater the diameter of induration, the stronger is that 1 piece of evidence.14 There are concerns over pertinence of TST positivity in patients from endemic areas with a high probability of being BCG vaccinated, as in our case; however, the effect on TST of BCG received in infancy is minimal, especially 10 years after vaccination. So, in such a scenario, a positive TST with indurations larger than 15 mm are more likely to be the result of TB infection than of BCG vaccination.15,16 Most forms of EPTB have a lower bacterial load than for pulmonary disease, being so-called pauci-bacillary forms. A relatively low proportion of cases have positive microscopy for AFB, and with the lower bacterial loads, even with rapid culture it takes longer to obtain positive cultures.17 Culture is a gold standard for TB diagnosis, but the long duration of about 4 to 6 weeks precludes delaying therapy and, hence, an appropriate treatment regimen should be started if histology and clinical picture are consistent with a diagnosis of TB.17 The culture can then be used for drug sensitivity testing, especially when suspecting multidrug-resistant strains.
OOOO Volume 116, Number 3
The newer technologies in TB diagnosis include serologic and molecular diagnosis, rapid culture techniques, and nucleic acid amplification tests. The commercially available serologic test kits are an attractive and early aid to tubercular diagnosis, although various studies have concluded that none of the antibody assays performed was good enough to replace microscopy.18-21 Various rapid culture methods have been developed based on radioactivity (BACTEC 460-TB), fluorescence (BACTEC MGIT 960), phage-based tests, and inverted microscopy leading to faster culture and sensitivity reports.22 The nucleic acid amplification test has been rapidly evolving for rapid and specific detection and identification of Mycobacterium tuberculosis. The evidence is limited concerning its performance in nonrespiratory specimens,23 with highly variable sensitivity and specificity reports as per various studies.19,23-30 Keeping in mind their high costs and requirement of instruments and trained manpower, use of such techniques in the foreseeable future will be very limited in disease-endemic countries, such as India, and should therefore be used in cases where there is a chance that the infection may be caused by a mycobacterium other than M. tuberculosis.31 The nucleic acid amplification test was not performed in our case. Evidence-based guidelines state that rapid diagnostic tests for M. tuberculosis complex identification should be conducted on biopsy material only if all the sample has been inappropriately placed in formalin, and AFB are visible on microscopy, whereas those on primary specimens should be used only if rapid confirmation of a TB diagnosis in a sputum smear–positive person would alter the person’s care, or before conducting a large contact-tracing initiative.28 Treatment of tuberculous osteomyelitis relies mainly on chemotherapy. Surgery is indicated only in cases with extensive destruction, presence of secondary infection, and intracranial involvement.8,32 Our patient completely recovered from the disease mainly from the anti-tubercular treatment (ATT) therapy with no surgical intervention. Radiological evidence of repair usually lags behind the clinical evidence of treatment.8 Prognosis is usually good if effective chemotherapy is available.
CONCLUSIONS Tuberculous osteomyelitis of zygoma is a rare condition. The key to diagnosis is mainly a high suspicion in the mind of the treating surgeon. The high suspicion, endemic area, contact history, positive Montoux test, radiological features, nonresponsiveness to conventional antibiotics, and histopathological examination
CASE REPORT Singh et al. e159
usually indicates the diagnosis. The condition is responsive to chemotherapy with surgery seldom being required. REFERENCES 1. Sethi A, Sethi D, Agarwal AK, Nigam S, Gupta A. Tubercular and chronic pyogenic osteomyelitis of cranio-facial bones: a retrospective analysis. J Laryngol Otol 2008;122:799-804. 2. Sethi A, Sareen D, Agarwal AK, Bansal R. Primary tuberculous osteomyelitis of zygoma. Int J Oral Maxillofac Surg 2006; 35:376-7. 3. Venkat Bhagirath P, Bhargavi Krishna A, Ashalata P, Reddy S. Primary tuberculous osteomyelitis of the mandible—a rare case report. Oral and Maxillofacial Pathology Journal 2011;2:117-22. 4. Ganguly PK. Radiology of bone and joint tuberculosis with special reference to tropical countries. London: Asia Publishing; 1963. p. 62. 5. Strauss DC. Tuberculosis of the flat bones of the vault of the skull. Surg Gynecol Obstet 1933;57:384-98. 6. Bhandari B, Mandowara SL, Joshi H. Tubercular osteomyelitis of skull. Indian J Pediatr 1981;48:113-5. 7. Sachdeva OP, Gulati SP, Kakar V, Sachdeva A, Arora B. Tuberculous osteomyelitis of zygoma. Trop Doct 1993;23:190-1. 8. Meher R, Garg A. Tubercular osteomyelitis of maxilla. Calicut Med J 2005;3:e4. 9. Malhotra R, Dinda AK, Bhan S. Tubercular osteitis of skull. Indian Pediatr 1993;30:1119-23. 10. Diagnostic standards and classification of tuberculosis in adults and children. Am J Respir Crit Care Med 2000;161:1376-95. 11. World Health Organization. Treatment of tuberculosis: guidelines for national programmes. Geneva; WHO; 1993:1-43. 12. Dadgarnia MH, Baradaranfar MH, Yazdani N, Kouhi A. Oropharyngeal tuberculosis: an unusual presentation. Acta Med Iranica 2008;46:521-24. 13. Tuberculosis Coalition for Technical Assistance. International standards for tuberculosis care (ISTC). 2nd ed. The Hague: Tuberculosis Coalition for Technical Assistance; 2009. 14. Harries AD, Maher D, Graham S. TB/HIV: a clinical manual. Stop TB Initiative. Geneva; World Health Organization; 2004. 15. Wang L, Turner MO, Elwood RK, Schulzer M, FitzGerald JM. A meta-analysis of the effect of Bacille Calmette Guerin vaccination on tuberculin skin test measurements. Thorax 2002; 57:804-9. 16. Farhat M, Greenaway C, Pai M, Menzies D. False-positive tuberculin skin tests: what is the absolute effect of BCG and non-tuberculous mycobacteria? Int J Tuberc Lung Dis 2006; 10:1192-204. 17. NICE clinical guideline 117; clinical diagnosis and management of tuberculosis, and measures for its prevention and control. Mid City Place, London; National Institute for Health & Clinical Excellence; March 2011. 18. Cunningham J. Paper presented at 36th Union World Conference on Lung Health of the International Union Against Tuberculosis and Lung Disease; 2005; Paris. 19. Dinnes J, Deeks J, Kunst H, Gibson A, Cummins E, Waugh N, et al. A systematic review of rapid diagnostic tests for the detection of tuberculosis infection. Health Technol Assess 2007; 11:1-196. 20. Steingart KR, Henry M, Laal S, Hopewell PC, Ramsay A, Menzies D, et al. A systematic review of commercial serological antibody detection tests for the diagnosis of extrapulmonary tuberculosis. Thorax 2007;62:911-8. 21. Steingart KR, Henry M, Laal S, Hopewell PC, Ramsay A, Menzies D, et al. Commercial serological antibody detection
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tests for the diagnosis of pulmonary tuberculosis: a systematic review. PLoS Med 2007;4:e202. Wood R. Challenges of TB diagnosis and TREATMENT in South Africa. Roche symposium, 3rd South African AIDS Conference, Durban, 5-8 June 2007. South Afr J HIV Med 2007;8: 44-8. Centers for Disease Control and Prevention. Updated guidelines for the use of nucleic acid amplification tests in the diagnosis of tuberculosis. MMWR Morb Mortal Wkly Rep 2009;58:7-10. Sarmiento OL, Weigle KA, Alexander J, Weber DJ, Miller WC. Assessment by meta-analysis of PCR for diagnosis of smearnegative pulmonary tuberculosis. J Clin Microbiol 2003;41: 3233-40. Pai M, Flores LL, Hubbard A, Riley LW, Colford JM Jr. Nucleic acid amplification tests in the diagnosis of tuberculous pleuritis: a systematic review and meta-analysis. BMC Infect Dis 2004;4:6. Pai M, Flores LL, Pai N, Hubbard A, Riley LW, Colford JM Jr. Diagnostic accuracy of nucleic acid amplification tests for tuberculous meningitis: a systematic review and meta-analysis. Lancet Infect Dis 2003;3:633-43. Flores LL, Pai M, Colford JM Jr, Riley LW. In-house nucleic acid amplification tests for the detection of Mycobacterium tuberculosis in sputum specimens: meta-analysis and meta-regression. BMC Microbiol 2005;5:55. Greco S, Girardi E, Navarra A, Saltini C. Current evidence on diagnostic accuracy of commercially based nucleic acid ampli-
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29.
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fication tests for the diagnosis of pulmonary tuberculosis. Thorax 2006;61:783-90. Daley P, Thomas S, Pai M. Nucleic acid amplification tests for the diagnosis of tuberculous lymphadenitis: a systematic review. Int J Tuberc Lung Dis 2007;11:1166-76. Ling DI, Flores LL, Riley LW, Pai M. Commercial nucleic-acid amplification tests for diagnosis of pulmonary tuberculosis in respiratory specimens: meta-analysis and meta-regression PLoS One 2008;3:e1536. Ichhpujani RL, Agarwal SP, Chauhan LS. Diagnostic needs and status of new diagnostic tools for tuberculosis; tuberculosis control in India. Tuberculosis Control India 2005;18:165-78. Ahmad Z, Masood I, Haque F, Abbas Z, Tamanna Z, Amin S. Tubercular osteomyelitis of zygomatic bones. J Indian Acad Clin Med 2007;8:276-7.
Reprint requests: Virendra Singh, MDS Professor and Head Department of Oral and Maxillofacial Surgery Government Dental College Pt. B.D. Sharma University of Health Sciences Rohtak-124001, Haryana, India [email protected]
Tuberculous osteomyelitis of zygoma: an unusual location Virendra Singh, MDS,a Shruti Khatana, MDS,b Pranav Gupta, MDS,b Amrish Bhagol, MDS,c and Anjali Narwal, MDS,d Haryana, India SHARMA UNIVERSITY OF HEALTH SCIENCES
Tubercular osteomyelitis of midfacial bones is extremely rare, although tuberculosis of long bones and the vertebral column is not uncommon. Because of the rare incidence, myriad presentation, and lack of specific symptoms, this condition presents a challenge in diagnosis and calls for acute clinical awareness. This article presents a case report of a 12-year-old girl with complaints of gradually increasing swelling lateral to and below her right eye for 4 months with pus discharge. She was treated with surgical curettage and 4-drug antitubercular therapy and responded with complete remission of the sinus. (Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:e156-e160)
Osteomyelitis of the craniofacial bones is an uncommon entity.1 The involvement of the bone in the middle third of the face is rare. The etiology most commonly arises from odontogenic sources or infected fracture sites and is usually pyogenic. Tubercular osteomyelitis of midfacial bones is extremely rare, although tuberculosis (TB) of long bones and vertebral column is not uncommon.2 Tubercular osteomyelitis is clinically and radiologically indistinguishable from pyogenic osteomyelitis and the 2 conditions can be differentiated only on the basis of histopathological evaluation of involved tissue.1 Because of the rare incidence, myriad presentation, and lack of specific symptoms, this condition presents a challenge in diagnosis and calls for acute clinical awareness. Representing a similar dilemma, we hereby report a case of tuberculous osteomyelitis of zygoma.
CASE REPORT A 12-year-old girl presented to the oral and maxillofacial surgery outpatient department of Government Dental College, Rohtak, India, with complaints of gradually increasing swelling lateral to and below her right eye for 4 months with pus discharge from the same for 15 days.
a
Professor and Head, Department of Oral and Maxillofacial Surgery, Government Dental College, Pt. B.D. Sharma University of Health Sciences, Haryana, India. b Resident, Department of Oral and Maxillofacial Surgery, Government Dental College, Pt. B.D. Sharma University of Health Sciences, Haryana, India. c Assistant Professor, Department of Oral and Maxillofacial Surgery, Government Dental College, Pt. B.D. Sharma University of Health Sciences, Haryana, India. d Assistant Professor, Department of Oral and Maxillofacial Pathology, Government Dental College, Pt. B.D. Sharma University of Health Sciences, Haryana, India. Received for publication Sep 20, 2011; returned for revision Dec 2, 2011; accepted for publication Dec 24, 2011. © 2013 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter doi:10.1016/j.oooo.2011.12.017
e156
A presumptive diagnosis of osteomyelitis was made and routine investigations and radiographs were ordered. Within a week of the presentation, the child complained of another fluctuant swelling on the right zygomatic prominence. Oral antibiotics from the penicillin group were started but no response was observed. Approximately 7 days later the child complained of redness of the right eye, which revealed conjunctival congestion, although the rest of the ophthalmologic examination was normal. On clinical examination, the swelling lateral and inferior to the right eye was firm and nontender on palpation with diffuse surrounding erythema. There was recurrent crusting and ulceration at the site, which on cleaning, revealed granulations at the margins and bed of the lesion. The overlying skin was adherent to the bone at certain areas. The other swelling that presented near the right zygomatic prominence measured 8 ⫻ 5 mm with fluctuation and discoloration that spontaneously burst and later persisted as a discharging sinus (Figure 1). The pus was sent for culture and sensitivity and did not show any growth/acid-fast bacilli (AFB)/sulfur granules. The patient had no systemic illness and was healthy otherwise. No history of trauma or previous infection was reported. There was no lymphadenopathy on examination. Routine blood investigations were normal. The erythrocyte sedimentation rate (ESR) was 50 mm/h in the first hour, hemoglobin was 12.0 g/dL, total leucocyte count (TLC) was 5500/mm3, differential leucocyte count (DLC) was P-58, L-36, M-6; enzyme-linked immunosorbent assay for HIV was negative; 48-hour Montoux reading was 27mm; and sputum for AFB was negative. On further inquiry, the patient’s father reported having been treated for pulmonary TB 1 year previously and was completely asymptomatic later. The child had no history of TB or any antitubercular therapy in past. The patient was Bacille Calmette-Guerin (BCG) vaccinated at infancy. Radiological investigations were done. Occipitomenton view revealed an ill-defined osteolytic lesion in the right zygomatic bone. Computed tomography (CT) scan of the face revealed few lytic lesions in relation to the right zygomatic bone with evidence of sclerosis and loculated subcutaneous soft tissue collection (Figure 2). Chest radiograph was normal.
OOOO Volume 116, Number 3
Fig. 1. Preoperative clinical photograph of the patient showing crusting and discharging sinus.
Fig. 2. CT scan of the face (coronal view) revealing few lytic lesions in relation to right zygomatic bone. The granulation tissue from the sinus was curetted intraorally under local anesthesia and the histopathology report revealed a caseating granulomatous lesion strongly suggestive of tuberculous osteomyelitis and was negative for AFB on special staining (Figure 3). The tissue specimen was also sent for culture, which was positive for mycobacterial growth by 5 to 6 weeks. The patient was put on a 4-drug antitubercular treatment (isoniazid, rifampicin, ethambutol, and pyrazinamide), and she responded with complete remission of the sinus. Her 6-month follow-up was uneventful (Figures 4 and 5).
DISCUSSION Tuberculosis is common in India and Southeast Asia, where the prevalence rate is about 4 in every 1000 people; 15% of the world’s tuberculous population resides in India.3
CASE REPORT Singh et al. e157
Fig. 3. Histopathological section showing chronic inflammatory infiltrate with giant cells (hematoxylin and eosin, original magnification ⫻10).
Fig. 4. Postoperative clinical photograph of the patient (6month follow-up).
Tuberculous osteomyelitis of the skull bones accounts for 0.1% to 3.7% of all cases of skeletal TB.4,5 Three-fourths of such cases were seen in patients younger than 20 years and half in those younger than 10 years.6 The present case was that of a 12-year-old girl. Most of the cases of TB of the skull are secondary to pulmonary TB7; however, there was no clinical or radiological evidence of pulmonary involvement in our case. Primary zygomatic TB is a rare condition and only a few cases have been reported.2 The presentation is nonspecific and usually confuses the diagnosis. A positive family history, endemic area, and infections not responding to conventional antibiotics should raise a suspicion. Raised ESR, highly positive Montoux, radiologic investigations, and histopa-
ORAL AND MAXILLOFACIAL SURGERY e158 Singh et al.
Fig. 5. Postoperative CT scan of the face (coronal view at 6-month follow-up).
thology are usually the key to diagnosis. A similar presentation was noted in our patient. Clinically, tuberculous osteomyelitis is characterized by lack of early symptoms.8 Appearance of a fluctuant swelling is usually the first symptom.8 Our case was characterized by 2 different appearances from the spectrum of presentations. The superior lesion was characterized by skin adhesion, erythema, crusting, and granulations, whereas the inferior lesion was typically fluctuant and spontaneously burst, producing a discharging sinus. The most common radiological presentation is a lytic lesion.8 The first demonstrable change in a small translucent area owing to decalcification is clinically undetectable at this stage. With the progress of the lesion, the decalcified areas increase in size with occurrence of bone destruction.2 It may present with a sclerotic border and later an osteoporotic edge.9 The dilemma is because radiologically the tuberculous osteomyelitis resembles nonspecific osteomyelitis.6 Our patient also presented with lytic lesions and sclerosis was visualized on the occipitomenton view and CT scan of the face. These findings can also be encountered in eosinophilic granuloma, congenital syphilis, or secondary malignant deposits.6 Such nonspecific presentation and overlapping symptoms make tuberculous osteomyelitis a diagnostic challenge, as in our case, where pyogenic osteomyelitis was the initial diagnosis. Moreover, extrapulmonary TB (EPTB) involves relatively inaccessible sites and because of the nature of the sites involved, fewer bacilli can cause much greater
OOOO September 2013
damage. The combination of small numbers of bacilli and inaccessible sites causes bacteriologic confirmation of a diagnosis to be more difficult, and invasive procedures are frequently required to establish a diagnosis.10 Diagnosis of EPTB is based on 1 culture-positive specimen from the extrapulmonary site, histologic evidence, or strong clinical evidence consistent with active EPTB disease followed by a medical officer’s decision to treat with a full course of anti-TB therapy.11 In our case, the histopathology report was highly suggestive of tuberculous osteomyelitis, although AFB was not demonstrated on special staining. The culture report was positive for mycobacterial growth by 5 to 6 weeks, although the chemotherapy had already been started based on histopathology and strong clinical suspicion. The good clinical response to chemotherapy obviated the need for any further diagnostics. Because of the relative scarcity of organisms within the tissue, the special stains successfully demonstrate the organism in only 27% to 60% cases.12 So, the absence of AFB in the tissue specimen does not rule out TB. The diagnostic pipeline for TB is replete with a multitude of tests but their relevance in terms of application and cost-effectiveness, especially in areas with a high burden of TB, such as India, Southeast Asia, and Africa, is yet to be established. Sputum smear microscopy, culture, biopsy and tubercular skin testing (TST) remain indispensable for tubercular diagnosis.13 A positive tuberculin test is only 1 piece of evidence in favor of the diagnosis of TB. The younger the child and the greater the diameter of induration, the stronger is that 1 piece of evidence.14 There are concerns over pertinence of TST positivity in patients from endemic areas with a high probability of being BCG vaccinated, as in our case; however, the effect on TST of BCG received in infancy is minimal, especially 10 years after vaccination. So, in such a scenario, a positive TST with indurations larger than 15 mm are more likely to be the result of TB infection than of BCG vaccination.15,16 Most forms of EPTB have a lower bacterial load than for pulmonary disease, being so-called pauci-bacillary forms. A relatively low proportion of cases have positive microscopy for AFB, and with the lower bacterial loads, even with rapid culture it takes longer to obtain positive cultures.17 Culture is a gold standard for TB diagnosis, but the long duration of about 4 to 6 weeks precludes delaying therapy and, hence, an appropriate treatment regimen should be started if histology and clinical picture are consistent with a diagnosis of TB.17 The culture can then be used for drug sensitivity testing, especially when suspecting multidrug-resistant strains.
OOOO Volume 116, Number 3
The newer technologies in TB diagnosis include serologic and molecular diagnosis, rapid culture techniques, and nucleic acid amplification tests. The commercially available serologic test kits are an attractive and early aid to tubercular diagnosis, although various studies have concluded that none of the antibody assays performed was good enough to replace microscopy.18-21 Various rapid culture methods have been developed based on radioactivity (BACTEC 460-TB), fluorescence (BACTEC MGIT 960), phage-based tests, and inverted microscopy leading to faster culture and sensitivity reports.22 The nucleic acid amplification test has been rapidly evolving for rapid and specific detection and identification of Mycobacterium tuberculosis. The evidence is limited concerning its performance in nonrespiratory specimens,23 with highly variable sensitivity and specificity reports as per various studies.19,23-30 Keeping in mind their high costs and requirement of instruments and trained manpower, use of such techniques in the foreseeable future will be very limited in disease-endemic countries, such as India, and should therefore be used in cases where there is a chance that the infection may be caused by a mycobacterium other than M. tuberculosis.31 The nucleic acid amplification test was not performed in our case. Evidence-based guidelines state that rapid diagnostic tests for M. tuberculosis complex identification should be conducted on biopsy material only if all the sample has been inappropriately placed in formalin, and AFB are visible on microscopy, whereas those on primary specimens should be used only if rapid confirmation of a TB diagnosis in a sputum smear–positive person would alter the person’s care, or before conducting a large contact-tracing initiative.28 Treatment of tuberculous osteomyelitis relies mainly on chemotherapy. Surgery is indicated only in cases with extensive destruction, presence of secondary infection, and intracranial involvement.8,32 Our patient completely recovered from the disease mainly from the anti-tubercular treatment (ATT) therapy with no surgical intervention. Radiological evidence of repair usually lags behind the clinical evidence of treatment.8 Prognosis is usually good if effective chemotherapy is available.
CONCLUSIONS Tuberculous osteomyelitis of zygoma is a rare condition. The key to diagnosis is mainly a high suspicion in the mind of the treating surgeon. The high suspicion, endemic area, contact history, positive Montoux test, radiological features, nonresponsiveness to conventional antibiotics, and histopathological examination
CASE REPORT Singh et al. e159
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Reprint requests: Virendra Singh, MDS Professor and Head Department of Oral and Maxillofacial Surgery Government Dental College Pt. B.D. Sharma University of Health Sciences Rohtak-124001, Haryana, India [email protected]