Primary nasal tuberculosis: a case report.

Abstract

During the past 2 decades, tuberculosis--both pulmonary and extrapulmonary--has re-emerged as a major health problem worldwide. Nasal tuberculosis--either primary or secondary to pulmonary tuberculosis or facial lupus--is rare, but it should be considered in the differential diagnosis of nasal granulomas. We describe a case of primary nasal tuberculosis in an adult male who presented with a polypoid lesion in one nasal cavity. The diagnosis was based on histopathology and the patient's successful response to antituberculous drug treatment. Given the rising incidence of tuberculosis, it is prudent that otolaryngologists remain cognizant of this infection as a potential cause of unusual lesions in the head and neck.

Introduction

Tuberculosis, a disease caused by acid- and alcohol-fast bacilli of the family Mycobacteriaceae (Mycobacterium tuberculosis, Mycobacterium bovis, Mycobacterium microti, and Mycobacterium africanum), is one of the earliest diseases known to affect humans. (1)

Tuberculous involvement of the nose is rare. When it does occur, it is usually secondary either to pulmonary tuberculosis or to lupus vulgaris of the facial skin. (2) Nasal tuberculosis was first described in 1761 by the Italian anatomy professor Giovanni Morgagni while reporting the autopsy findings of a young man with pulmonary tuberculosis who had ulcerations of the nose, soft palate, and nasopharynx. (3)

Primary tuberculosis of the nose is also rare. The first case of primary tuberculosis of the upper respiratory tract and nose was presented to the Pathological Society of London by Clarke in 1852. (3) Later in the 18th century, reviews published by Herzog described 20 cases of primary nasal tuberculosis among 80 cases of nasal tuberculosis overall. (3) In a review of the 20th-century medical literature published in 1997, Butt found only 35 cases of nasal tuberculosis. (4) Our independent review added eight other recently reported cases. (5-11)

In this article, we report a new case of primary nasal tuberculosis, and we review the literature relevant to the incidence, investigation, and treatment of this condition.

Case report

Our patient was a 55-year-old man who was known to have had noninsulin-dependent adult-onset diabetes mellitus for 8 years. He was monitored regularly, and his diabetes was well controlled on oral medication.

On March 6, 2002, he was referred by his general practitioner to the Department of Otorhinolaryngology at our institution for evaluation of a 4-week history of right nasal obstruction and occasional epistaxis. His general practitioner had prescribed a course of antibiotic and antihistamine therapy for nonspecific rhinitis, but treatment did not relieve his symptoms.

Office endoscopy of the nose revealed the presence of a reddish polypoid lesion on the anterior edge of the inferior turbinate in the right nasal cavity (figure 1). The adjacent mucosa over the turbinate and the mucosa over the septum were thick and bled when touched. The lesion was biopsied under topical anesthesia; minimal bleeding was noted. A diagnosis of granulomatous disease of the nose was tendered.

[FIGURE 1 OMITTED]

The patient exhibited no clinical evidence of any systemic discasc. Hematologic tests revealed the following: (1) his erythrocyte sedimentation rate was 20 mm/hr by Westergren's method (normal: <15 mm/hr), (2) he had no antibodies to human imnmnodeficiency virus, and (3) his glycosylated hemoglobin [A.sub.1c] level was 8% of the total, which suggested adequate control of his diabetes. Results of renal and liver function tests were normal. His Mantoux' test elicited a significant reaction: 20 mm after 72 hr. Three sputum smears were negative for acid-fast bacilli, as were subsequent cultures.

Findings on computed tomography of the paranasal sinuses were normal except for hypertrophy of the turbinate on the right side; there was no evidence of any lesion in the paranasal sinuses. Findings oil chest x-rays were normal.

Biopsy analysis revealed that the caseating granulomatous lesion was made up of epithelioid cells, lymphocytes, and a few giant cells. The stroma contained many lymphocytes and a few mucosal glands (figure 2). Special staining for fungus and leprid bacilli was negative. A few acid-fast bacilli were noted on Ziehl-Neelsen staining. These findings suggested a diagnosis of tuberculosis. Because no other focus of the disease was identified, a diagnosis of primary nasal tuberculosis was reached.

[FIGURE 2 OMITTED]

In accordance with the Ministry of Health of Oman's guidelines for the treatment of extrapulmonary tuberculosis, the patient was administered isoniazid, rifampin, and pyrazinamide for 2 months, followed by isoniazid and rifampin for 4 months. (12) Treatment resulted in a rapid resolution of symptoms and a regression of the nasal lesion. Culture of the specimen was subsequently reported as negative. The histopathologic picture and the clinical response to therapy confirmed the diagnosis of primary nasal tuberculosis.

The patient underwent regular follow-up, including comprehensive clinical and otolaryngologic examination with nasal endoscopy, and he remained disease-free at 2 years. Systematic contact tracing did not reveal any tuberculosis in his family or immediate associates, although his occupation as a taxi driver might have put him at risk for occasional contact.

Discussion

Antituberculous chemotherapy and public health measures led to a dramatic decline in the incidence of tuberculosis by the middle of the 20th century. However, since 1986, a steady and progressive increase in the number of cases has been reported worldwide. (13) Although tuberculosis is more common in developing nations, an increase has been noted even in developed countries such as the United States. (14)

Acquired immunodeficiency syndrome (AIDS), which progressively destroys the cell-mediated immunity required to combat tuberculosis, is one of the primary factors responsible for the resurgence of tuberculosis. Other factors implicated are inadequate public health systems and crowded and substandard living conditions. Poverty and inadequate access to health care place elderly homeless populations and those in drug treatment centers and correctional facilities at greatest risk. (1,14)

The incidence of extrapulmonary tuberculosis has also increased. Extrapulmonary tuberculosis represents a diagnostic challenge to specialists in the involved anatomic area. This disease is rarely infectious, and patients typically have bacterial counts much lower than do those who have cavitary pulmonary disease. (14) Patients with extrapulmonary tuberculosis account for only 15% of patients with tuberculosis, but 70% of patients with AIDS and tuberculosis. (14) It should be noted that tuberculosis is often difficult to diagnose in patients with AIDS because their radiographic findings are often atypical, they lack granuloma formation, and their purified-protein-derivative skin tests are negative. (1,13)

Nasal tuberculosis can be caused by either a pulmonary disease or a retrograde involvement of the nose by lupus vulgaris of the facial skin. In only a few cases does it represent a primary infection. Primary disease is caused by inhalation of infected particles or traumatic digital inoculation. The infrequency of nasal tuberculosis might be explained by the protection afforded by ciliary movement, the bactericidal action of nasal secretions, and the filtering provided by the nasal vibrissae. It has also been postulated that the nasal mucosa is inherently resistant to mycobacterial growth.

Reported cases of nasal tuberculosis have occurred in patients between the ages of 20 and 84 years (median: mid-40s). The condition occurs more often in women than men. The presenting symptoms in their order of frequency are nasal obstruction, nasal discharge, nasal discomfort, epistaxis, crusting, eye watering, postnasal discharge, recurrent nasal polyps, and ulceration. Lesions may be ulcerative, infiltrative, or proliferative, and most cases are unilateral. The most common sites of involvement are the cartilaginous septum and the inferior turbinate. (4)

The diagnosis is difficult to establish because the symptoms and signs of the various conditions that make up the differential diagnosis are similar and nonspecific (table). (14,15) A definitive diagnosis is made by identifying or isolating tuberculous bacilli from tissue removed during biopsy or surgery. Nasal secretions and swab specimens have a very low yield and should not be used to rule out this condition. On histology, both caseating and noncaseating granulomas have been described. Biopsies of noncaseating granulomas are confusing histologically, and these cases are often misdiagnosed as Wegener's granuloma. The numbers of Langhans' giant cells and epithelioid cells are believed to be greater in tuberculosis than in other granulomas. (3) Acid-fast bacilli may be demonstrated on Ziehl-Neelsen staining or on auramine-rhodamine staining under fluorescent microscopy. Cultures of involved tissues are more often positive than are smears. A portion of any biopsy specimen should be sent for culture, along with a notification of the types of organisms that are suspected. (14)

The roles of polymerase chain reaction assays, DNA probes, and high-performance liquid chromatography are limited by their cost, lack of availability, and lack of sensitivity. These modalities are currently recommended for species identification and to detect primary resistance to drugs. (1,13)

Some patients with nasal tuberculosis subsequently develop pulmonary tuberculosis and/or extrapulmonary tuberculosis in adjacent sites such as the larynx. (4) Therefore, the physician should look for these concomitant manifestations during both the initial work-up and subsequent follow-up.

The treatment of nasal tuberculosis should follow the general guidelines established for the treatment of extrapulmonary tuberculosis, although variations in the choice of medications may be necessary according to the availability of drugs and the local incidence of resistant strains. These guidelines usually call for a short initial bactericidal regimen (often three or more drugs) followed by a prolonged sterilizing regimen (at least two drugs). (1)

The case of nasal tuberculosis described in this report was noteworthy because the patient was apparently healthy and his immune status had not been demonstrably compromised. The presence of both nasal tuberculosis and diabetes may have been coincidental as opposed to causal.

Given current trends in the incidence of tuberculosis, it would be prudent for otolaryngologists to remain cognizant of tuberculosis as a potential entity when encountering an unusual lesion in the head and neck. (14) Although nasal tuberculosis is rare, it should be considered in the differential diagnosis of chronic nasal symptoms and granulomatous lesions of the nose. The nasal granuloma in our patient was diagnosed as primary nasal tuberculosis on the basis of histology and the clinical response to antituberculous medication.

Table. Classification of nasal granulomas

Infectious       Mycobacterial: Tuberculosis (human,
                 bovine, or avian); atypical tuberculo-
                 sis; leprosy
                 Bacterial: Rhinoscleroma
                 Treponemal: Syphilis; yaws
                 Fungal: Mucormycosis; aspergillosis;
                 blastomycosis; histoplasmosis;
                 coccidiomycosis; rhinosporidiosis
                 Parasitic: Leishmaniasis
Noninfectious    Wegener's granulomatosis
                 Sarcoidosis
                 Inclusion granuloma (silicosis;
                 berylliosis)
                 Foreign-body retention
Malignant        Lethal midline granuloma
Nonspecific

Acknowledgments

The authors thank the Director General of Health Services, the Superintendent for Planning and Training in the Al Dhahira Region, and the Medical Superintendent at Al Buraimi Hospital for permitting and facilitating this report. We also gratefully acknowledge the assistance of Prof. Euan Scrimgeour, Head of Infectious Diseases, and Prof. Anand Datey and Dr. V. Nirmala of the Department of Pathology at the Sultan Qaboos University Hospital.

References

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(4.) Butt AA. Nasal tuberculosis in the 20th century. Am J Med Sci 1997;313:332-5.

(5.) Blanco Aparicio M, Verea Hernando H, Pombo F. Tuberculosis of the nasal fossa manifested by a polypoid mass. J Otolaryngol 1995;24:317-18.

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(7.) Rao S, Rau PV, Sahoo RC, et al. Primary nasal tuberculosis. Tuber Lung Dis 1992;73:305.

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(9.) Choi YC, Park YS, Jeon EJ, Song SH. The disappeared disease: Tuberculosis of the nasal septum. Rhinology 2000;38:90-2.

(10.) Hup AK, Haitjema T, de Kuijper G. Primary nasal tuberculosis. Rhinology 2001;39:47-8.

(11.) Batra K, Chaudhary N, Motwani G, Rai AK. An unusual case of primary nasal tuberculosis with epistaxis and epilepsy. Ear Nose Throat J 2002;81:842-4.

(12.) Treatment Schedule in Ministry of Health, Sultanate of Oman. TB Manual. 3rd ed. 1998, Section 6:16-17.

(13.) Johnson IJM, Soames Jr, Marshall HF. Nasal tuberculosis--an increasing problem? J Laryngol Otol 1995;109:326-7.

(14.) Goguen LA, Karmody CS. Nasal tuberculosis. Otolaryngol Head Neck Surg 1995;113:131-5.

(15.) Hughes RG, Drake Lee A. Nasal manifestations of granulomatous disease. Hosp Med 2001;2:417-21.

Ravi C. Nayar, MS, DLO (RCS), DCCF (Paris)

Juma Al Kaabi, MBBS, MRCP (U.K.)

Kanchanmala Ghorpade, MD (Pathol) (Bombay)

>From the Department of Otorhinolaryngology (Dr. Nayar), the Department of Medicine (Dr. Al Kaabi), and the Department of Pathology (Dr. Ghorpade), Al Buraimi Hospital, Sultanate of Oman.

Reprint requests: Dr. Ravi C. Nayar, Head, Department of Otorhinolaryngology, Ibri Regional Referral Hospital, PO Box 46, Postal Code 516, Al Dhahira Region, Sultanate of Oman. Phone: [phone], ext. 222; fax: [phone]; e-mail [e-mail]