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Cervical and Mediastinal Tuberculous Lymphadenitis Associated With Tracheal Lesion

Shikama, Yusuke M.D.; Nakajima, Hiroaki M.D.; Mizuno, Tetuo M.D.; Ueno, Kana M.D.; Adachi, Mitsuru M.D.

Brief Report
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Tuberculous mediastinal lymphadenitis without pulmonary infiltrate in an adult is rare in Japan and in western countries. We report a 37-year-old man with cervical and mediastinal tuberculous lymphadenitis. Contrast-enhanced computed tomography revealed multiple enlarged lymph nodes with central low attenuation and peripheral rim enhancement. Flexible bronchoscopic examination demonstrated a tracheal polypoid lesion on the right wall. These lesions disappeared after 10 months of antituberculosis chemotherapy. Furthermore, soluble interleukin-2 receptor seemed to be a useful parameter of the treatment.

First Department of Internal Medicine, Showa University School of Medicine, Tokyo, Japan

Address reprint requests to Dr. Yusuke Shikama, 21–2 Enokigaoka Aoba-ku Yokohama-shi, Kanagawa, Japan.

sIL-2R, soluble interleukin-2 receptor.

The increasing incidence of tuberculosis has been attributed to several causes including acquired immune deficiency syndrome and other important medical problems in many countries. 1 Intrathoracic lymphadenitis with pulmonary infiltration is recognized as a characteristic manifestation of primary tuberculosis, especially in children. However, tuberculous mediastinal lymphadenitis without demonstrable pulmonary involvement in adults is rare in Japan and in western countries. 2–4 We report an adult case of cervical and mediastinal tuberculous lymphadenitis with tracheal lesion and an elevated serum soluble interleukin-2 receptor (sIL-2R) level.

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CASE REPORT

A 37-year-old man was referred to our institute for examination of a large tumor on the left side of his neck. The patient reported shoulder stiffness for 3 months before the first visit to our institute. He had been in excellent health until then and worked as a television director. He smoked one pack of cigarettes daily. He had no history of risk factors for human immunodeficiency virus infection. On physical examination, the patient appeared to be in good health, with a blood pressure of 126/82 mmHg, a temperature of 36.6°C, a pulse of 72, and a respiratory rate of 15. The large tumor, which measured 10 × 8 × 6 cm on the left side of his neck, was movable, elastic, and hard, and its edge was clear. Breath sounds were clear and no heart murmurs were audible. Abdominal findings were normal. Laboratory test results indicated a white blood cell count of 8,700 cells/μL; erythrocyte sedimentation rate, 81 mm/hour; C-reactive protein, 4.5 mg/dL; and sIL-2R in the blood, 851 U/mL. Other blood chemical and enzyme values were normal. Tumor markers such as carcinoembryonic antigen, sialyl SSGA-1 antigen, squamous cell carcinoma-related antigen, and pro-gastrin releasing peptide were within normal ranges, and the patient's serum angiotensin-converting enzyme level was normal. Serum test for antinuclear antibodies was positive, and the test for antihuman immunodeficiency virus antibody was negative. Urinalysis was normal. A tuberculin test (purified protein derivatives) was positive. A chest radiograph taken on the first visit showed widening of the mediastinum without parenchymal lung infiltration. Computed tomographic scans of the chest revealed large, multiple lymphadenopathies on the left neck and mediastinum with central or multifocal low-density areas and peripheral rim enhancement (Fig. 1). However, no calcification was detectable. Gallium-67 scan showed increased radioactivity in the enlarged cervical and mediastinal lymph nodes and the cecum (Fig. 2). Examination of sputum revealed neither malignancy nor tuberculosis. Needle aspiration samples from the cervical tumor showed yellow pus, and cytologic evaluation revealed no malignant cells. Acid-fast stain was negative and routine culture was negative. However, polymerase chain reaction for tuberculosis of the aspiration samples was positive, suggesting lymphadenitis resulting from tuberculosis. Two weeks later the patient was admitted to our hospital and antituberculosis chemotherapy—rifampicin, isoniazid, ethambutol, and pyrazinamide—was started. Flexible bronchoscopy was performed and demonstrated a trachea polypoid lesion of 8 mm elevated from the right wall, 5 cm proximal to the carina (Fig. 3A). At that time, no attempt was made to perform a biopsy because we were concerned about the spread of tuberculosis to the airway. Only bronchial lavage was performed, and malignant cells and acid-fast bacilli were not detectable. On the fifth day after admission, the cervical mass was ruptured and yellow pus exuded from it. Mycobacterium tuberculosis was identified on the 5-week culture of aspiration materials from the patient's first visit to our hospital. The patient's condition was good, with no clinical symptoms while in the hospital. After 2 months of intensive chemotherapy, antituberculosis therapy was continued at our clinic. The mediastinal lymph node swelling decreased gradually and was not detectable by chest computed tomography at 10 months. The shape and size of the tracheal lesion showed no remarkable changes during the 5 months of observation. However, after 10 months of treatment the tracheal lesion disappeared, leaving a scar at the base (Fig 3B). The elevated serum sIL-2R level and erythrocyte sedimentation rate declined gradually and normalized within 2 months (Fig. 4). After 1 year of treatment, antituberculosis therapy was discontinued and the patient was monitored at our clinic.

FIG. 1.

FIG. 1.

FIG. 2.

FIG. 2.

FIG. 3.

FIG. 3.

FIG. 4.

FIG. 4.

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DISCUSSION

Tuberculous mediastinal lymphadenopathy in adults is infrequent in Japan and in western countries. The frequency of hilar and mediastinal lymphadenitis is less than 5% in Japan and 4.4% in the United States among tuberculosis patients, with a female-to-male predominance of 2.8:1. 4,5 Tuberculous mediastinal lymphadenitis without pulmonary lesion is much more rare in adults. 2,3 In contrast, the incidence of tuberculosis in Pakistan and India is approximately 20 times higher than in western countries. It is recognized that tuberculous mediastinal lymphadenopathy in these countries is more frequent than in the West. 6 Tuberculosis is one of the important causes of mediastinal lymphadenopathy in these countries. It is likely that tuberculous mediastinal lymphadenitis in adults is usually a manifestation of primary tuberculosis, as it is in children; however, reactivation of a latent nodal focus probably accounts for some cases. 2 As evidenced by our patient, the right tracheobronchial region has been reported to be the most commonly affected site. 2,7 Previous studies demonstrated that right intrathoracic lymphatic vessels combine and form a main truncus, whereas left lymphatic vessels lead individually to the thoracic duct. 8 Because there is considerable left-to-right crossover of lymphatic drainage but much less from right to left, right tracheobronchial adenopathy is common. 2 Contrast-enhanced computed tomographic findings of nodules with central low attenuation and peripheral rim enhancement corresponding pathologically to caseation or liquefaction necrosis and granulation tissue with inflammatory hypervascularity respectively are indicators of active tuberculous disease. 9,10 Enlarged lymph nodes with a low attenuation center are also seen in other diseases such as metastatic malignancy and pyogenic infection, whereas lymphomatous adenopathy characteristically shows homogeneous attenuation. 11 Gallium-67 scanning is a useful method for the detection of active inflammatory lesions and is sensitive for determining the localization of extrapulmonary tuberculosis. 12 The tracheal lesion was not detectable by conventional computed tomography. Obviously, bronchoscopic biopsy is necessary for the diagnosis of the tracheal polypoid lesion such as adenoma, carcinoma, inflammatory polyp, benign tumor, and amiloidoma. 13 In our patient the tracheal lesion had a smooth surface and looked like an inflammatory polyp. A histologic approach was reserved because of the chance of infection spreading to other patients. The tracheal polypoid lesion disappeared after 10 months of antituberculosis treatment, suggesting that this lesion was associated with chronic inflammation of tuberculous lymphadenopathy. Many patients with bronchial tuberculosis have few acid-fast bacilli in the sputum and have tuberculous lesions confined predominantly to the submucosa. 14 Perforation of the hilar lymph nodes into the bronchi 15 and spread to the intercostal chest wall have been reported previously. 4 Careful observation is recommended, particularly because of initial exacerbation induced by antituberculosis chemotherapy. In our patient, no serious complications or drug side effects were observed.

sIL-2R is a marker of T-lymphocyte activation, and T lymphocytes are essential in immunity against tuberculosis. 16 As reported previously, sIL-2R is elevated in the serum of patients with active pulmonary tuberculosis and declines gradually after antituberculosis therapy. 16,17 These findings were observed in our patient. sIL-2R was a useful parameter to determine the effects of treatment.

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REFERENCES

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Keywords:

Mediastinal lymphadenitis; Tracheal lesion; Tuberculosis; sIL-2R

© 2002 Lippincott Williams & Wilkins, Inc.