Coexistence of a tuberculous bronchoesophageal fistula and intracranial tuberculosis in an immunocompetent patient.

To the Editor: Extrapulmonary involvement of tuberculosis increases with the rise of the incidence of immunocompromised host, especially in acquired immunodeficiency syndrome, and it can occur even in immunocompetent patients. (1) We report an immunocompetent case with coexistence of a tuberculous bronchoesophageal fistula and intracranial tuberculosis, successfully managed by antituberculous chemotherapy alone.

An 18-year-old female presented with a two-day history of left hemiparesis. There was low-grade fever and cough since two months before admission. One month later, paroxysms of cough with choking following ingestion of liquids occurred. The patient was febrile (38.6[degrees]C), respiratory rate was 18/min and blood pressure was 108/68 mm Hg. Chest x-ray showed right lower lung infiltrations and a widened mediastinum. Computed tomography (CT) of the brain without contrast enhancement revealed a hypodense lesion at the posterior limb of the right internal capsule and increased local vascularities after contrast enhancement in this area. Human immunodeficiency virus (HIV) test was negative.

On the 2nd day of admission, CT of the chest revealed right lower lobe infiltrations and enlarged lymph nodes in the right hilar, right paratracheal and subcarinal regions. Empiric treatment with antituberculous agents was started through a nasogastric feeding tube. Bronchoscopy disclosed an elevated lesion over the proximal portion of the right main bronchus and biopsy specimens showed chronic inflammation. Sputum smears were negative for acidfast bacilli, and sputum cytology was also negative for malignancy. Magnetic resonance (MR) imaging of the brain showed one lesion in the right temporal lobe with low signal on T1-weighted images and high signal on T2-weighted images and another in the subcortical white matter of left frontal lobe with high signal on T2-weighted images. Gadolinium-enhanced T1-weighted MR imaging demonstrated multiple diffusely enhancing lesions in the basal cisterns, bilateral frontal, right temporal and left parietal areas (Fig. 1). A barium swallow performed on the 12th hospital day demonstrated a fistulous communication between the mid-esophagus and the right main bronchus (Fig. 2). Esophagoscopy revealed a large ulcer with a central fistulous opening, 28 cm from the incisor. Biopsy specimens from the ulcer margin showed caseating granulomas containing Langhans giant cells. The patient improved and was discharged on the 22nd hospital day. Follow-up esophagogram after antituberculous treatment of one and a half months' duration showed disappearance of the bronchoesophageal fistula. Occasional mild headaches were noted during 1-year treatment course.

[FIGURE 1 OMITTED]

[FIGURE 2 OMITTED]

Communication between the esophagus and the tracheobronchial tree is an unusual clinical condition, which may be either congenital (2) or acquired. (3) In the acquired form of esophagorespiratory fistula, it is considered to be secondary to malignancies, trauma or infections. Mycobacterium tuberculosis was the most common pathogen causing infectious fistulas, and the fistulous tracts were mostly noted at the right main bronchus, (4) as in our case. Intracranial tuberculosis, causing a granulomatous inflammatory reaction that involves the meninges and/or the brain parenchyma, results from hematogenous spread of Mycobacterium tuberculosis from a preexisting focus elsewhere in the body. The appearance of intracranial tuberculous lesions on CT or MR imaging depends on the maturity and type of lesion.

Isolation of tubercle bacilli and tissue proof of caseating granulomata histologically are sometimes difficult, especially in patients with extrapulmonary tuberculosis. Although tubercle bacilli was not found in our case, the diagnosis of tuberculosis was based on a combination of clinical, radiological, endoscopic, histologic features and response to the chemotherapy.

Extrapulmonary tuberculosis has been on a rise with the increase in the incidence of HIV infection. However, in endemic regions of tuberculosis, one should have a strong index of suspicion of extrapulmonary tuberculosis even in HIV-negative patients. Extrapulmonary tuberculosis is potentially curable and needs an efficient diagnostic workup so that treatment may be initiated rapidly.

Hsin-Hui Chiu, MD

Joe Hergens Li, MD

Department of Internal Medicine

Jackson Ching-Tzer Lin, MD, PhD

Department of Radiology

Yao-Wen Liu, MD

Department of Pathology

Kuo General Hospital

Tainan, Taiwan

Yeun Tarl Fresner Ng Jao, MD

Department of Internal Medicine

Tainan Municipal Hospital

Tainan, Taiwan

References

1. Louie E, Rice LB, Holzman RS. Tuberculosis in non-Haitian patients with acquired immunodeficiency syndrome. Chest 1986;90:542-545.

2. Risher WH, Arensman RM, Ochsner JL. Congenital bronchoesophageal fistula. Ann Thorac Surg 1990;49:500-505.

3. Mellins RB. Acquired fistula between the esophagus and the respiratory tract; report of a case, review of the literature and discussion of the pathogenesis. N Engl J Med 1952;246:896-901.

4. Asnis DS, Saltzman HP, Giron JA. Bronchoesophageal fistula due to multidrug-resistant tuberculosis in a patient infected with human immunodeficiency virus. Clin Infect Dis 1995;21:1061-1062.Letters to the Editor