From the Departments of Pediatrics and *E.N.T. G.G.S. Medical College, Faridkot 151 203, Punjab, India.

Correspondence to: Dr. Gurmeet Kaur, c/o Dr. A.S. Sethi, Jaitu Road, Kotkapura 151 204, Punjab, India.

Manuscript received: March 13, 2002;
Initial review completed: April 3, 2002;
Revision accepted: September 27, 2002.

An 11-year-old female child presented with high grade intermittent fever and cough for a duration of 6-7 months and hoarseness of voice for 6 months. Skiagram of the chest showed evidence of miliary mottling. Direct laryngoscopic examination revealed inflammatory swelling over left vocal cord. The biopsy of the swelling showed chronic granulomatous lesion. Patient improved remarkably with anti-tubercular therapy.

Key words: Larygeal, tuberculosis.

 

With the advent of effective chemotherapy, involvement of larynx in tuberculosis is now a rare phenomenon. In the pre-antibiotic era it was a common complication of advanced pulmonary, active cavitatory disease, as the tubercle bacilli involved the larynx as the infective sputum was coughed up(1). We present here a case of miliary tuberculosis with laryngeal involvement because of its rarity.

An 11-year-old female child presented with the complaints of high grade, intermittent fever and cough with yellowish, non-foul smelling expectoration for the last 6-7 months and hoarseness of voice of 6 weeks duration. She had received BCG and there was no history of hemoptysis, dyspnea, dysphagia, loss of appetite or significant loss of weight. There was a history of close contact with a neighbour suffering from chronic pulmonary tuberculosis. She had received several courses of antiboitics without any response.

Physical examination revealed a sick - looking, moderately anemic, febrile child. Her pulse rate, respiratory rate and blood pressure were 180/min, 38/min and 120/70 mm of Hg respectively. Throat examination was normal. Auscultation of the chest revealed diffuse bilateral, scattered rhonchi and coarse crepitations. Hemogram showed Hb - 6.5 g/dL, TLC - 8000/cumm, DLC - P62 L35 M1 E2 and ESR - 120 mm in 1st hour. Mantoux test and sputum for AFB were negative. Skiagram of the chest showed evidence of miliary mottling and cystic lesions in the right lower zone and left upper zone. Direct laryngoscopic examination done under anesthesia revealed an inflammatory swelling mainly over the left vocal cord. The biopsy material from the involved area revealed chronic granulomatous lesions consisting of a collection of epitheloid cells surrounded by lymphocytes.

The patient was put on appropriate anti-tubercular drugs. She became afebrile, gained 2 kg weight and her hoarseness improved gradually. At 6 weeks follow up she is doing well and her voice is almost normal.

Classically, laryngeal tuberculosis is described in middle-aged males usually with concurrent far advanced pulmonary tuberculosis, in whom its clinical presentation is similar to that of laryngeal carcinoma(2,3). There are very few case reports of laryngeal tuberculosis in children(2-5), the youngest reported case being 14 months old(6). Hoarseness, which was the main presenting symptom in our case, does not include laryngeal tuberculosis in its differential diagnosis because of its rarity. The common causes of hoarseness include foreign body, trauma, congenital laryngeal anomalies, acute laryngitis and weakness of the laryngeal muscles(7). However, against a background of symptomatology usually associated with tuberculosis (viz. fever, cough, weight loss and night sweats), varying degrees of hoarseness with or without painful dysphagia are suggestive of laryngeal tuberculosis(2). Hoarseness may be produced by lesions on the cord or by cordal immobility. Another uncommon symptom of laryngeal tuberculosis is stridor(4,5,8). An association with tuberculous enteritis has also been reported(3,8) but both were not seen in our patient.

Laryngeal tuberculosis can occur regardless of the immunization status and result of tuberculin test. Some reported patients have been previously immunized with BCG(4) and others have had a negative Mantoux test(3,8). Our patient had also received BCG and her Mantoux test was negative.

The pathogenesis of laryngeal tuberculosis may be either primary or secondary. Primary infection of the larynx occurs when there is absence of pulmonary disease and the organism directly infects the mucosa via aerosolized particles resulting in the formation of a granuloma(5). Secondary infection of the larynx may occur by direct spread as in active, advanced, cavitatory pulmonary tuberculosis where the highly infectious sputum is expectorated up the tracheobronchial tree (bronchogenic theory). In this case the infection usually remains intralaryngeal, mainly around the true vocal cords. The lesions are ulcerative and granulomatous. Healing occurs with minimal fibrosis. Hematogenous or lymphatic spread to the larynx occurs less frequently and is more likely in patients having minimal pulmonary involvment or miliary tuberculosis. Hematogenous spread is more common to the extrinsic larynx (i.e. epiglottis, arytenoids and aryepiglottic folds) although true cords may be involved. Diffuse vascular necrosis and severe painful edema often involves large areas of the larynx. Healing is usually accompanied by progressive fibrosis and stenosis may result(3,5,8). The adult form of laryngeal tuberculosis is invariably secondary to direct spread(5). In contrast, children usually have no evidence of pulmonary disease and their chest-x-rays at presentation are usually normal(3,5,8). However, our patient presented with prolonged chest symptoms, miliary mottling on X-ray chest and a localized tubercular granulomatous lesion on left vocal cord suggesting an "adult" form of the disease. There were no confluent cavitatory lesions in any segment which could suggest a bronchogenic spread of the infection to the larynx. Although miliary tuberculosis is usually associated with hematogenous dissemination and extensive involvement of the larynx with residual stenosis, our patient had a pseudotumoral form of laryngeal tuberculosis with complete healing as evidenced by disappearance of hoarseness within 6 weeks of starting antitubercular therapy. Vocal cord immobility due to fibrosis as is seen in hematogenous spread would have led to persistent hoarseness(9).

In recent years the clinical and morphological aspects of tubercular laryngitis have changed significantly from what they were before chemotherapy and the most common clinical form is now pseudotumoral tuberculosis(10). Since majority of the patients have no evidence of chronic disease, they may be diagnosed as juvenile laryngeal papillomatosis, the most common laryngeal tumor in children(8,10).

A definitive diagnosis of laryngeal tuberculosis is made by isolating M.tuberculosis from a biopsy specimen, but this may not always be possible(3). It was not done in our case and the diagnosis was evident from coexistent miliary tuberculosis, laryngoscopy findings and histopathology report. Remarkable response to anti-tubercular therapy further confirmed the diagnosis.

Contributors: GK worked up the patient and drafted the manuscript which was supervised and edited by KS. TLP performed the direct laryngoscopy and biopsy and helped in analyzing its findings. GK shall act as the guarantor.

Funding: None.

Competing interests: None stated.

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