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Primary tuberculosis of the tongue: report of three cases
BJOM-133.QXD
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BRITISH
British Journal of Oral and Maxillofacial Surgery (2001) 39, 402–403 © 2001 The British Association of Oral and Maxillofacial Surgeons doi: 10.1054/bjom.2001.0663, available online at http://www.idealibrary.com on
Journal of Oral and Maxillofacial Surgery
Primary tuberculosis of the tongue: report of three cases E. M. Iype,* K. Ramdas,† M. Pandey,* K. Jayasree,‡ G. Thomas,§ P. Sebastian,* M. K. Nair† *Division of Surgical Oncology; †Radiation Oncology; ‡Pathology; §Community Oncology, Regional Cancer Centre, Trivandrum, India SUMMARY. Tuberculosis of the oral cavity is uncommon and poses a diagnostic challenge. We report three patients with primary tuberculosis of the tongue, all of whom responded well to treatment and were cured. © 2001 The British Association of Oral and Maxillofacial Surgeons
The response was excellent and the tongue lesion healed well. The patient is disease-free after 2 years.
INTRODUCTION Tuberculosis (TB) is a chronic infectious granulomatous disease caused by acid fast Mycobacterium tuberculosis and less commonly by M. bovis and other atypical mycobacteria. Extrapulmonary involvement in TB is rare, accounting for about 10–15% of all cases. Involvement of the oral cavity is documented in about 0.2–1.5% of the cases, and is generally secondary to pulmonary TB; primary involvement is exceedingly rare. The tongue is the most commonly affected site.1–7 The other sites in the oral cavity are lips,5,6 cheek,7,8 soft palate,9 uvula, and gingival and alveolar mucosa. Recently there has been a resurgence of interest because of the association of TB with HIV infection. We report three cases of primary TB of the tongue seen at the Regional Cancer Centre, Trivandrum, India.
CASE 2 A 47-year-old man, a chronic smoker and alcoholic, presented with a non-healing ulcer on the tongue of one month’s duration. Examination showed a 32 cm indurated ulcer with undermined edges, on the anterior third of the tongue involving the tip and right lateral border. The ulcer was not tender on palpation and its base was covered by a greyish-white slough. Bilateral submandibular nodes were palpable. His haematological and biochemical measurements were within the reference range and chest radiograph was within normal limits. Sputum showed no acid-fast bacilli. Histopathological examination of the incisional biopsy specimen showed caseating granulomatous lesions. With a diagnosis of primary TB of the tongue, the patient was treated with a course of four antituberculous drugs. Subsequent follow-up showed excellent clinical improvement and the patient is disease-free after 5 years.
CASE 1 A 52-year-old deaf and dumb man, a chronic smoker, was referred with a non-healing, painful, intraoral ulcer and intolerance to hot and spicy food of a month’s duration. There was a 41 cm linear ulcer on the dorsum of the tongue extending from the middle third to the base of the tongue. The ulcer was erythematous with an irregular undermined border and the base was covered by a greyish-white slough. Haematological and biochemical measurements were within the reference ranges except for an increased erythrocyte sedimentation rate (ESR), which was 30 mm in the first hour. Chest X-ray picture was normal. Sputum showed no acid fast bacilli. A punch biopsy of the ulcer showed multiple granulomatous foci composed of epithelioid cells and Langhans giant cells in the subepithelial region. Small foci of necrosis was also noted. The patient was treated with antituberculous drugs consisting of rifampicin, pyrazinamide, ethambutol, and isoniazid.
CASE 3 A 51-year-old man, a chronic smoker, was referred with a nonhealing intraoral ulcer. Examination showed a 33 cm ulcer on the left lateral border of the tongue. The ulcer was not tender on palpation and there was minimal induration and irregular undermined borders. The routine haematological, and biochemical investigations were within the reference ranges and chest X-ray was normal. Histopathological examination of the incision biopsy specimen showed caseating granulomatous lesions composed of epithelioid cells and Langhans giant cells. Acid first bacilli were seen on Ziehl–Neelson staining. The patient was given four-drug antituberculous treatment. Complete remission of the tongue lesion was noted after 6 weeks and the patient is disease-free after 6 years. 402
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Primary TB of the tongue
DISCUSSION TB of the oral cavity may be either primary or secondary.4 Secondary TB is more common among older patients and is usually a complication of pulmonary disease.4 Primary TB, on the other hand, is unusual, is seen in younger patients, and may be associated with cervical lymphadenopathy. The mechanism of primary inoculation is not known. However, it is thought that the mycobacterium is inoculated directly into the oral mucosa.10 The intact oral mucous membrane presents a natural resistance to invasion by mycobacterium. This has been attributed to the cleansing action of saliva, the presence of salivary enzymes, tissue antibodies, oral saprophytes, and the thickness of the protective epithelial covering. Any break or loss of this natural barrier, which may be the result of trauma, inflammatory conditions, tooth extraction, or poor oral hygiene, may provide a route of entry for the organism. TB is an important differential diagnosis of chronic oral ulcers. They should be differentiated from traumatic ulcers, aphthous ulcers, actinomycosis, syphilitic ulcers, lymphoma and metastatic lesions.11 As with other tuberculous lesions of the head and neck, they can resemble carcinoma or the two can coexist. This emphasizes the importance of an early biopsy and bacteriological tests to differentiate between the lesions. REFERENCES 1. Gupta KB. Tuberculosis of tongue: a case report. Lung India 1998; 16: 32–33. 2. Jaward J, EL Znebi F. Primary lingual tuberculosis : a case report. J Laryngol Otol 1996; 110: 1778–1780. 3. Hashimoto Y, Tanika H. Primary tuberculosis of tongue: report of a case. J Oral Maxillofac Surg 1989; 47: 744–746.
403
4. Hock-Leiw Eng, Shin Yu Lu, Chuang Hwa Yang et al. Oral tuberculosis. Oral Surg Oral Med Oral Pathol 1996; 81: 415– 420. 5. Macfarlane TW, Samaranayake LP. Clinical oral microbiology. London: Butterworth, 1989; 112–115. 6. Rao TV, Satyanarayanan CV, Sundareshwar B et al. Unusual form of tuberculosis of lip. J Oral Surg 1977; 35: 595–596. 7. Mitchell RG, Brown RM, Massland EA. An unusual tuberculosis lesion of the oral mucous membrane. Br J Oral Surg 1966; 4: 1–5. 8. Haddad NM, Zaytoun GM, Hadi U. Tuberculosis of the soft palate: an unusual presentation of oral tuberculosis. Otolaryngol Head Neck Surg 1987; 97: 91–99. 9. Mani NJ. Tuberculosis initially diagnosed by symptomatic oral lesion: report of three cases. J Oral Med 1985; 40: 39–42. 10. Rauch MD, Friedman E. Systemic tuberculosis initially seen as an oral ulceration: report of a case. J Oral Surg 1978; 36: 387–389. 11. Prabhu SR, Daftary DK, Dholakia HM. Tuberculous ulcer of the tongue: report of a case. J Oral Surg 1978; 36: 384–386.
The Authors Elizabeth M. Iype DNB Division of Surgical Oncology K. Ramdas MD Division of Radiation Oncology Manoj Pandey MS Division of Surgical Oncology K. Jayasree MD Division of Pathology Gigi Thomas MDS Division of Community Oncology Paul Sebastian MS Division of Surgical Oncology M. Krishnan Nair FRCR Division of Radiation Oncology Regional Cancer Centre Trivandrum, India Correspondence and requests for offprints to: Manoj Pandey MS, Assistant Professor, Surgical Oncology, Regional Cancer Centre, Medical College P.O., Thiruvananthapuram, Kerala 695 011, India. Tel: +91471 443 667; Fax:+91471 447 454; E-mail: [email protected] or [email protected] Accepted 2 April 2001 Published online 5 July 2001
9/20/01 8:50 AM
Page 402
BRITISH
British Journal of Oral and Maxillofacial Surgery (2001) 39, 402–403 © 2001 The British Association of Oral and Maxillofacial Surgeons doi: 10.1054/bjom.2001.0663, available online at http://www.idealibrary.com on
Journal of Oral and Maxillofacial Surgery
Primary tuberculosis of the tongue: report of three cases E. M. Iype,* K. Ramdas,† M. Pandey,* K. Jayasree,‡ G. Thomas,§ P. Sebastian,* M. K. Nair† *Division of Surgical Oncology; †Radiation Oncology; ‡Pathology; §Community Oncology, Regional Cancer Centre, Trivandrum, India SUMMARY. Tuberculosis of the oral cavity is uncommon and poses a diagnostic challenge. We report three patients with primary tuberculosis of the tongue, all of whom responded well to treatment and were cured. © 2001 The British Association of Oral and Maxillofacial Surgeons
The response was excellent and the tongue lesion healed well. The patient is disease-free after 2 years.
INTRODUCTION Tuberculosis (TB) is a chronic infectious granulomatous disease caused by acid fast Mycobacterium tuberculosis and less commonly by M. bovis and other atypical mycobacteria. Extrapulmonary involvement in TB is rare, accounting for about 10–15% of all cases. Involvement of the oral cavity is documented in about 0.2–1.5% of the cases, and is generally secondary to pulmonary TB; primary involvement is exceedingly rare. The tongue is the most commonly affected site.1–7 The other sites in the oral cavity are lips,5,6 cheek,7,8 soft palate,9 uvula, and gingival and alveolar mucosa. Recently there has been a resurgence of interest because of the association of TB with HIV infection. We report three cases of primary TB of the tongue seen at the Regional Cancer Centre, Trivandrum, India.
CASE 2 A 47-year-old man, a chronic smoker and alcoholic, presented with a non-healing ulcer on the tongue of one month’s duration. Examination showed a 32 cm indurated ulcer with undermined edges, on the anterior third of the tongue involving the tip and right lateral border. The ulcer was not tender on palpation and its base was covered by a greyish-white slough. Bilateral submandibular nodes were palpable. His haematological and biochemical measurements were within the reference range and chest radiograph was within normal limits. Sputum showed no acid-fast bacilli. Histopathological examination of the incisional biopsy specimen showed caseating granulomatous lesions. With a diagnosis of primary TB of the tongue, the patient was treated with a course of four antituberculous drugs. Subsequent follow-up showed excellent clinical improvement and the patient is disease-free after 5 years.
CASE 1 A 52-year-old deaf and dumb man, a chronic smoker, was referred with a non-healing, painful, intraoral ulcer and intolerance to hot and spicy food of a month’s duration. There was a 41 cm linear ulcer on the dorsum of the tongue extending from the middle third to the base of the tongue. The ulcer was erythematous with an irregular undermined border and the base was covered by a greyish-white slough. Haematological and biochemical measurements were within the reference ranges except for an increased erythrocyte sedimentation rate (ESR), which was 30 mm in the first hour. Chest X-ray picture was normal. Sputum showed no acid fast bacilli. A punch biopsy of the ulcer showed multiple granulomatous foci composed of epithelioid cells and Langhans giant cells in the subepithelial region. Small foci of necrosis was also noted. The patient was treated with antituberculous drugs consisting of rifampicin, pyrazinamide, ethambutol, and isoniazid.
CASE 3 A 51-year-old man, a chronic smoker, was referred with a nonhealing intraoral ulcer. Examination showed a 33 cm ulcer on the left lateral border of the tongue. The ulcer was not tender on palpation and there was minimal induration and irregular undermined borders. The routine haematological, and biochemical investigations were within the reference ranges and chest X-ray was normal. Histopathological examination of the incision biopsy specimen showed caseating granulomatous lesions composed of epithelioid cells and Langhans giant cells. Acid first bacilli were seen on Ziehl–Neelson staining. The patient was given four-drug antituberculous treatment. Complete remission of the tongue lesion was noted after 6 weeks and the patient is disease-free after 6 years. 402
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9/20/01 8:50 AM
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Primary TB of the tongue
DISCUSSION TB of the oral cavity may be either primary or secondary.4 Secondary TB is more common among older patients and is usually a complication of pulmonary disease.4 Primary TB, on the other hand, is unusual, is seen in younger patients, and may be associated with cervical lymphadenopathy. The mechanism of primary inoculation is not known. However, it is thought that the mycobacterium is inoculated directly into the oral mucosa.10 The intact oral mucous membrane presents a natural resistance to invasion by mycobacterium. This has been attributed to the cleansing action of saliva, the presence of salivary enzymes, tissue antibodies, oral saprophytes, and the thickness of the protective epithelial covering. Any break or loss of this natural barrier, which may be the result of trauma, inflammatory conditions, tooth extraction, or poor oral hygiene, may provide a route of entry for the organism. TB is an important differential diagnosis of chronic oral ulcers. They should be differentiated from traumatic ulcers, aphthous ulcers, actinomycosis, syphilitic ulcers, lymphoma and metastatic lesions.11 As with other tuberculous lesions of the head and neck, they can resemble carcinoma or the two can coexist. This emphasizes the importance of an early biopsy and bacteriological tests to differentiate between the lesions. REFERENCES 1. Gupta KB. Tuberculosis of tongue: a case report. Lung India 1998; 16: 32–33. 2. Jaward J, EL Znebi F. Primary lingual tuberculosis : a case report. J Laryngol Otol 1996; 110: 1778–1780. 3. Hashimoto Y, Tanika H. Primary tuberculosis of tongue: report of a case. J Oral Maxillofac Surg 1989; 47: 744–746.
403
4. Hock-Leiw Eng, Shin Yu Lu, Chuang Hwa Yang et al. Oral tuberculosis. Oral Surg Oral Med Oral Pathol 1996; 81: 415– 420. 5. Macfarlane TW, Samaranayake LP. Clinical oral microbiology. London: Butterworth, 1989; 112–115. 6. Rao TV, Satyanarayanan CV, Sundareshwar B et al. Unusual form of tuberculosis of lip. J Oral Surg 1977; 35: 595–596. 7. Mitchell RG, Brown RM, Massland EA. An unusual tuberculosis lesion of the oral mucous membrane. Br J Oral Surg 1966; 4: 1–5. 8. Haddad NM, Zaytoun GM, Hadi U. Tuberculosis of the soft palate: an unusual presentation of oral tuberculosis. Otolaryngol Head Neck Surg 1987; 97: 91–99. 9. Mani NJ. Tuberculosis initially diagnosed by symptomatic oral lesion: report of three cases. J Oral Med 1985; 40: 39–42. 10. Rauch MD, Friedman E. Systemic tuberculosis initially seen as an oral ulceration: report of a case. J Oral Surg 1978; 36: 387–389. 11. Prabhu SR, Daftary DK, Dholakia HM. Tuberculous ulcer of the tongue: report of a case. J Oral Surg 1978; 36: 384–386.
The Authors Elizabeth M. Iype DNB Division of Surgical Oncology K. Ramdas MD Division of Radiation Oncology Manoj Pandey MS Division of Surgical Oncology K. Jayasree MD Division of Pathology Gigi Thomas MDS Division of Community Oncology Paul Sebastian MS Division of Surgical Oncology M. Krishnan Nair FRCR Division of Radiation Oncology Regional Cancer Centre Trivandrum, India Correspondence and requests for offprints to: Manoj Pandey MS, Assistant Professor, Surgical Oncology, Regional Cancer Centre, Medical College P.O., Thiruvananthapuram, Kerala 695 011, India. Tel: +91471 443 667; Fax:+91471 447 454; E-mail: [email protected] or [email protected] Accepted 2 April 2001 Published online 5 July 2001