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Tuberculous Esophagitis with Aortic Aneurysm Fistula
Tuberculous Esophagitis with Aortic Aneurysm Fistula Frank P. Catinella, M.D., and C. Frederick Kittle, M.D.
ABSTRACT A patient with treated pulmonary tuberculosis and a thoracic aortic aneurysm was seen with a onemonth history of dysphagia. Barium swallow revealed a mass in the lower esophagus and extravasation of contrast material into the mediastinurn. Endoscopy and biopsy specimens showed acid-fast organisms. The patient was treated with antituberculous drug therapy but bled massively from the gastrointestinal tract and died. Autopsy revealed an aortoesophageal fistula at the level of the thoracic aneurysm. Histopathological study confirmed that this rare tuberculous lesion of the esophagus caused the fistula. Tuberculosis of the esophagus is rare. Most often it is secondary to tuberculous lesions at other sites (lungs, mediastinum, or larynx), although primary submucosal lesions in the esophagus have been noted. The lesions may be ulcerative, hyperplastic, or granular and often mimic carcinoma [l-41. Involvement of neighboring structures may occur with tracheobronchial fistula, tuberculous aortitis, and aneurysm formation [5-91. Tuberculous aneurysms frequently rupture into the gastrointestinal tract, but fistulas from tuberculosis of the gastrointestinal tract extending into a nontuberculous aorta are less common [7-91. We report a tuberculous esophageal fistula to an atherosclerotic thoracic aortic aneurysm. An 87-year-old woman was seen with a one-month history of dysphagia. Her medical history included resection of an abdominal aortic aneurysm, a stable thoracic aortic aneurysm, hyperthyroidism, and pulmonary tuberculosis, for which she had undergone a full course of drug therapy. Physical findings included tubercular breath sounds at the right lung base, a grade 4/6 systolic cardiac murmur at the left lower sternal border radiating to the left axilla, a grade 2/6 early diastolic murmur at the lower left sternal border, and cachexia. A nontender, 5cm, pulsatile mass was present in the upper abdomen. Admission laboratory values were remarkable for mild anemia (hematocrit, 37%)and a slightly elevated level of alkaline phosphatase (138 U/L). Chest roentgenogram showed hilar and pulmonary calcifications, a descending thoracic aortic aneurysm that was unchanged when From the Department of Cardiovascular-Thoracic Surgery, RushPresbyterian-St. Luke’s Medical Center, Rush Medical College, Chicago, IL. Accepted for publication Apr 22, 1987. Address reprint requests to Dr. Kittle, Rush-Presbyterian-St. Luke’s Med Center, 1753 West Congress Pkwy, Chicago, IL 60612.
compared with roentgenograms done two years previously, and progressive cardiomegaly. A preliminary diagnosis of benign esophageal stricture rather than carcinoma of the esophagus was considered. Barium contrast study of the esophagus showed a lesion in the distal third of the esophagus with extravasation of contrast material outside the lumen of the esophagus above the gastroesophageal junction (Fig 1). Computed tomographic scan of the chest demonstrated a large aneurysm of the descending thoracic aorta (Fig 2). A small collection of contrast material was juxtaposed between the heart and the thoracic aorta. There was no evidence of mediastinal adenopathy or of other parenchymal soft tissue abnormalities. Esophagoscopy revealed an obstructing, fungating esophageal mass in the distal third of the esophagus; multiple biopsy specimens were obtained. Microscopic examination of these specimens revealed squamous epithelium with submucosal acute and chronic inflammation and areas of granulomatous inflammation containing acid-fast bacilli. A gastrostomy tube was placed, and triple-drug therapy (ethambutol, rifampin, and streptomycin) was begun. Subsequently, sputum and gastric aspirate cultures from the time of admissjon became positive for Mycobucteriurn tuberculosis. On the thirty-third hospital day, she bled massively from the gastrointestinal tract and died. At autopsy, a 10-cm saccular atheroscleroticaneurysm of the descending thoracic aorta was found filled with a laminar mural thrombus. A fistulous tract extended into the esophagus approximately 1.5 cm above the gastroesophageal junction. The entire gastrointestinal tract was filled with fresh blood. Microscopically, granulomas were found throughout the wall of the esophagus at the fistulous site, but not in the wall of the aorta. The mediastinal nodes contained old, calcified granulomas; there was no evidence of active inflammation elsewhere.
Comment Tuberculous esophagitis was first confirmed at autopsy in 1837 by Denonvilliers and later, in 1877, by Zenker and Ziemssen [2]. The first clinical diagnosis by esophagoscopy was made by Schrotter in 1907 [2]. The common routes of infection are swallowing sputum that contains acid-fast bacilli or erosion of bronchi or lymph nodes with active infection into the esophagus [2]. Its rarity suggests a possible increased resistance to infection by the esophageal mucosa. Dysphagia is usually the presenting symptom, and three forms of the disease are evident-hypertrophic, granular, and ulcerative. The last form is most common, while the granular form is the rarest. The ulcerative form may be seen as solitary or multiple ulcers, not unlike tuberculous ulceration elsewhere in the body. It has been observed at every level
87 Ann Thorac Surg 45:87-88, Jan 1988. Copyright 0 1988 by The Society of Thoracic Surgeons
88 The Annals of Thoracic Surgery Vol 45 No 1 January 1988
Fig 1 . Barium upper gastrointestinal series. Note extravasation of contrast media from the lumen of the esophagus into the mediastinum. Also shown are a traction diverticulum and a cakified hilar node.
but is most common just above the tracheal bifurcation. The ulcers are typically superficial, but they may penetrate into the muscular layers with fistula formation. Resolution of the disease has been noted with proper antituberculous drug treatment [l,3, 41. Relentless progression of the disease generally occurs despite medical therapy, with the complications of traction diverticula, esophagobronchial fistula, esophagotracheal fistula, esophagopleural fistula, esophagocutaneous fistula, recurrent nerve paralysis, and malignant change [l].Surgical intervention has produced disappointing results [5, 61. Aortic involvement with tuberculosis is relatively rare, although over 100 cases have been documented since 1882 [8]. A focus of aortitis becomes established, and the aortic wall undergoes destruction. Ensuing complications depend on the extent and rapidity of the inflammatory process. Thus, perforation with massive hemorrhage and formation of true and false aneurysms, dissecting aneurysms, and arterial mycotic emboli have been noted [7-91. Analysis of these cases reveals that perforation and aneurysm formation are the most common complications, usually terminating in fatal hemorrhage. Spread of the infection to the aorta is usually by direct extension from a contiguous pathological process (tuberculous lymphadenitis, pericarditis, empyema, spondylitis, or paravertebral abscess). To our knowledge, this report is the first instance of direct fistulization from a tuberculous esophagus into a nontuberculous atherosclerotic aneurysm of the aorta.
References
Fig 2 . Computed tomographic chest scan revealing a 10 x 10-cm aneuysm of the descending thoracic aorta and an abnormal collection of contrast material in a plane between the posterior aspect of the heart and the aneurysm.
1. Fahmy AP, Guind R, Farid A: Tuberculosis of the esophagus. Thorax 24:154, 1969 2. Lockard L D Esophageal tuberculosis: a critical review. Laryngoscope 23:561, 1913 3. Sauge PE, Grundy A: Oesophageal tuberculosis: an unusual cause of dysphagia. Br J Radio1 571153, 1984 4. Seivewright N, Feehally J, Wicks ACB: Primary tuberculosis of the esophagus. Am J Gastroenterol 79:842, 1984 5. Yacoub MH, You KD, Kittle CF: Non-malignant tracheobroncho-oesophageal fistula in an adult. Br J Dis Chest 67161, 1973 6. Pecora D V Tuberculous fistula of the esophagus. J Thorac Surg 36:53, 1958 7. Stiefel J W Rupture of a tuberculous aneurysm of the aorta. AMA Arch Pathol 65:506, 1958 8. OLeary M, Nollet DJ, Blomberg DJ: Rupture of a tuberculous pseudoaneurysm of the innominate artery into the trachea and esophagus: report of a case and review of the literature. Hum Pathol 8:458, 1977 9. Braman R, Napoli V M Exsanguination from esophageal rupture of an atherosclerotic aortic aneurysm in a patient with treated tuberculosis. South Med J 72:892, 1979
ABSTRACT A patient with treated pulmonary tuberculosis and a thoracic aortic aneurysm was seen with a onemonth history of dysphagia. Barium swallow revealed a mass in the lower esophagus and extravasation of contrast material into the mediastinurn. Endoscopy and biopsy specimens showed acid-fast organisms. The patient was treated with antituberculous drug therapy but bled massively from the gastrointestinal tract and died. Autopsy revealed an aortoesophageal fistula at the level of the thoracic aneurysm. Histopathological study confirmed that this rare tuberculous lesion of the esophagus caused the fistula. Tuberculosis of the esophagus is rare. Most often it is secondary to tuberculous lesions at other sites (lungs, mediastinum, or larynx), although primary submucosal lesions in the esophagus have been noted. The lesions may be ulcerative, hyperplastic, or granular and often mimic carcinoma [l-41. Involvement of neighboring structures may occur with tracheobronchial fistula, tuberculous aortitis, and aneurysm formation [5-91. Tuberculous aneurysms frequently rupture into the gastrointestinal tract, but fistulas from tuberculosis of the gastrointestinal tract extending into a nontuberculous aorta are less common [7-91. We report a tuberculous esophageal fistula to an atherosclerotic thoracic aortic aneurysm. An 87-year-old woman was seen with a one-month history of dysphagia. Her medical history included resection of an abdominal aortic aneurysm, a stable thoracic aortic aneurysm, hyperthyroidism, and pulmonary tuberculosis, for which she had undergone a full course of drug therapy. Physical findings included tubercular breath sounds at the right lung base, a grade 4/6 systolic cardiac murmur at the left lower sternal border radiating to the left axilla, a grade 2/6 early diastolic murmur at the lower left sternal border, and cachexia. A nontender, 5cm, pulsatile mass was present in the upper abdomen. Admission laboratory values were remarkable for mild anemia (hematocrit, 37%)and a slightly elevated level of alkaline phosphatase (138 U/L). Chest roentgenogram showed hilar and pulmonary calcifications, a descending thoracic aortic aneurysm that was unchanged when From the Department of Cardiovascular-Thoracic Surgery, RushPresbyterian-St. Luke’s Medical Center, Rush Medical College, Chicago, IL. Accepted for publication Apr 22, 1987. Address reprint requests to Dr. Kittle, Rush-Presbyterian-St. Luke’s Med Center, 1753 West Congress Pkwy, Chicago, IL 60612.
compared with roentgenograms done two years previously, and progressive cardiomegaly. A preliminary diagnosis of benign esophageal stricture rather than carcinoma of the esophagus was considered. Barium contrast study of the esophagus showed a lesion in the distal third of the esophagus with extravasation of contrast material outside the lumen of the esophagus above the gastroesophageal junction (Fig 1). Computed tomographic scan of the chest demonstrated a large aneurysm of the descending thoracic aorta (Fig 2). A small collection of contrast material was juxtaposed between the heart and the thoracic aorta. There was no evidence of mediastinal adenopathy or of other parenchymal soft tissue abnormalities. Esophagoscopy revealed an obstructing, fungating esophageal mass in the distal third of the esophagus; multiple biopsy specimens were obtained. Microscopic examination of these specimens revealed squamous epithelium with submucosal acute and chronic inflammation and areas of granulomatous inflammation containing acid-fast bacilli. A gastrostomy tube was placed, and triple-drug therapy (ethambutol, rifampin, and streptomycin) was begun. Subsequently, sputum and gastric aspirate cultures from the time of admissjon became positive for Mycobucteriurn tuberculosis. On the thirty-third hospital day, she bled massively from the gastrointestinal tract and died. At autopsy, a 10-cm saccular atheroscleroticaneurysm of the descending thoracic aorta was found filled with a laminar mural thrombus. A fistulous tract extended into the esophagus approximately 1.5 cm above the gastroesophageal junction. The entire gastrointestinal tract was filled with fresh blood. Microscopically, granulomas were found throughout the wall of the esophagus at the fistulous site, but not in the wall of the aorta. The mediastinal nodes contained old, calcified granulomas; there was no evidence of active inflammation elsewhere.
Comment Tuberculous esophagitis was first confirmed at autopsy in 1837 by Denonvilliers and later, in 1877, by Zenker and Ziemssen [2]. The first clinical diagnosis by esophagoscopy was made by Schrotter in 1907 [2]. The common routes of infection are swallowing sputum that contains acid-fast bacilli or erosion of bronchi or lymph nodes with active infection into the esophagus [2]. Its rarity suggests a possible increased resistance to infection by the esophageal mucosa. Dysphagia is usually the presenting symptom, and three forms of the disease are evident-hypertrophic, granular, and ulcerative. The last form is most common, while the granular form is the rarest. The ulcerative form may be seen as solitary or multiple ulcers, not unlike tuberculous ulceration elsewhere in the body. It has been observed at every level
87 Ann Thorac Surg 45:87-88, Jan 1988. Copyright 0 1988 by The Society of Thoracic Surgeons
88 The Annals of Thoracic Surgery Vol 45 No 1 January 1988
Fig 1 . Barium upper gastrointestinal series. Note extravasation of contrast media from the lumen of the esophagus into the mediastinum. Also shown are a traction diverticulum and a cakified hilar node.
but is most common just above the tracheal bifurcation. The ulcers are typically superficial, but they may penetrate into the muscular layers with fistula formation. Resolution of the disease has been noted with proper antituberculous drug treatment [l,3, 41. Relentless progression of the disease generally occurs despite medical therapy, with the complications of traction diverticula, esophagobronchial fistula, esophagotracheal fistula, esophagopleural fistula, esophagocutaneous fistula, recurrent nerve paralysis, and malignant change [l].Surgical intervention has produced disappointing results [5, 61. Aortic involvement with tuberculosis is relatively rare, although over 100 cases have been documented since 1882 [8]. A focus of aortitis becomes established, and the aortic wall undergoes destruction. Ensuing complications depend on the extent and rapidity of the inflammatory process. Thus, perforation with massive hemorrhage and formation of true and false aneurysms, dissecting aneurysms, and arterial mycotic emboli have been noted [7-91. Analysis of these cases reveals that perforation and aneurysm formation are the most common complications, usually terminating in fatal hemorrhage. Spread of the infection to the aorta is usually by direct extension from a contiguous pathological process (tuberculous lymphadenitis, pericarditis, empyema, spondylitis, or paravertebral abscess). To our knowledge, this report is the first instance of direct fistulization from a tuberculous esophagus into a nontuberculous atherosclerotic aneurysm of the aorta.
References
Fig 2 . Computed tomographic chest scan revealing a 10 x 10-cm aneuysm of the descending thoracic aorta and an abnormal collection of contrast material in a plane between the posterior aspect of the heart and the aneurysm.
1. Fahmy AP, Guind R, Farid A: Tuberculosis of the esophagus. Thorax 24:154, 1969 2. Lockard L D Esophageal tuberculosis: a critical review. Laryngoscope 23:561, 1913 3. Sauge PE, Grundy A: Oesophageal tuberculosis: an unusual cause of dysphagia. Br J Radio1 571153, 1984 4. Seivewright N, Feehally J, Wicks ACB: Primary tuberculosis of the esophagus. Am J Gastroenterol 79:842, 1984 5. Yacoub MH, You KD, Kittle CF: Non-malignant tracheobroncho-oesophageal fistula in an adult. Br J Dis Chest 67161, 1973 6. Pecora D V Tuberculous fistula of the esophagus. J Thorac Surg 36:53, 1958 7. Stiefel J W Rupture of a tuberculous aneurysm of the aorta. AMA Arch Pathol 65:506, 1958 8. OLeary M, Nollet DJ, Blomberg DJ: Rupture of a tuberculous pseudoaneurysm of the innominate artery into the trachea and esophagus: report of a case and review of the literature. Hum Pathol 8:458, 1977 9. Braman R, Napoli V M Exsanguination from esophageal rupture of an atherosclerotic aortic aneurysm in a patient with treated tuberculosis. South Med J 72:892, 1979