å¡ CASE REPORT å¡ Recurrent Hemoptysis due to Aortobronchopulmonary Fistula of False Aortic Aneurysm Associated with Repair of Rupture of the Sinus of Valsalva Akihiro Ishida, Kazuyoshi Sakai, Masayoshi Ajioka, Tetsuo Hiramatsu, Yoshihito Nakashima, Motoaki Hoshino* and Teiji Asakura* A 54-year-old man presented with recurrent hemoptysis of one year duration. Computed tomography (CT) and magnetic resonance imaging (MRI) demonstrated a saccular aneurysm of the ascending aorta. The aneurysm was intraoperatively found to have formed on the superior surface of the site of aortotomy suture placed during previous repair of rupture of the sinus of Valsalva and to have a fistulous communication to the lung. CT and MRI were very useful in (Internal Medicine 1043-1046, 1992) the diagnosis of the31: aneurysm as the cause of hemoptysis. Key words: aortotomy, computed tomography, magnetic resonance imaging, aortography
Intro duction
negative, and tuberculin skin test was doubtful. Chest radiograph showed granular and reticular shadows at False aneurysm is a rare complication of aortotomy per bilateral lower lung fields and a questionable contour formed during cardiosurgery. Although hemoptysis due to adjacent to the right upper cardiac silhouette (Fig. 1). Bronchoscopy revealed no source of hemoptysis, and aortobronchopulmonary fistula of the aneurysm is usually bronchography revealed no specific findings for bron fatal if untreated, recurrent hemoptysis as the sole symp chiectasis. CT showed a 4 x 4 cm saccular shadow anterior tom is most unusual. We report such a case in which com and to the right of the ascending aorta. This shadow was puted tomography (CT) and magnetic resonance imaging partially enhanced to the same intensity as the aortic lumen, (MRI) were very useful in the diagnosis of the aneurysm suggesting a saccular aneurysm many clots (Fig. 2A). and in which surgery was successful. MRI also clearly showed that thewith shadow had a communica Case Report tion with the lumen of the ascending aorta (Fig. 2B). Aortography at the root of the aorta revealed that a small A 54-year-old man was referred to our hospital for re quantity of contrast medium had leaked out of the current hemoptysis of one year duration. The patient had ascending aorta, and insertion of the catheter into the undergone repair of rupture of the sinus of Valsalva aneurysm was then attempted. Although manual injection of a small amount of contrast medium into the aneurysm sustained inhea had traffic accident six years prior to admission. Thereafter, suffered mediastinitis and osteomyelitis confirmed that this shadow was a false aneurysm arising of the sternum, and the rectus abdominal muscle had been from the ascending aorta, no aortobronchopulmonary transplanted in a sternotomy chest wound. The patient was asymptomatic, and neither heart Intraoperatively, aneurysm found communicationsthe were detectedwas (Fig. 3). to be mostly filled with thrombotic material, and a 10 mm diameter rent murmurs nor abnormal breathing sounds were noted. Pulse in the aorta on the superior surface of the site of previous was regular at 66/min, and blood pressure was 140/86 aortotomy suture was visible. Removal of the thrombi mmHg. Hemoglobin concentration was 13.4 g/dl, and disclosed a 1 mm diameter fistula on the right floor of the leukocyte count was 5.9 x lOVmm3. C-reactive protein was aneurysm. Artificial ventilation demonstrated leakage of air negative. Examination of the sputum showed no malignant into the aneurysm via the fistula, confirming its comcells or tubercle bacilli. Blood serology for syphilis was From the Department of Internal Medicine and *the Department of Cardiovascular Surgery, Tosei General Hospital, Seto Received for publication April 17, 1991; Accepted for publication May 6, 1992 Reprint requests should be addressed to Dr. Akihiro Ishida, the Department of Internal Medicine, Tosei General Hospital, 160 Nishioiwak Seto489, Japan Internal
Medicine
Vol. 31, No. 8 (August
1992)
1043
Ishida
et al munication with the lung. Histologic examination showed that the aneurysm was of the false variety (Fig. 4). The patient's postoperative course was uneventful.
Fig. 1. Chest radiograph shows a questionable contour adjacent Fig. 3. Injection of contrast medium into the aneurysm reveals it to the right upper cardiac silhouette (arrow). to be a false aneurysm arising from the ascending aorta.
Fig. 2. A) CT at the level of bifurcation of pulmonary artery shows a saccular shadow filled with thrombus (left arrow) with enhance ment equaling that of the aortic lumen (right arrow). B) MRI shows this shadow to have a communication with the aortic lumen (arrow 1 044
Internal
Medicine
Vol. 31,
No. 8 (August
1992)
Hemoptysis from Postoperative
Aneurysm
pressure, in a patient with chronic hemoptysis has been reported (7). Fortunately, in the present case, low pressure manual injection of contrast medium into the aneurysm caused no massive hemoptysis as a result of the dislodge ment of the clot obstructing the fistula. Injection of even a small amount of contrast medium provided satisfactory images. Frequently, aortography is essential to formulate the decisions regarding surgical procedures. Bronchoscopy is helpful only in the diagnosis of an endobronchial tumor as the source of the bleeding and, for the same reason as in aortography, there is a risk of lethal hemorr hage associated with its use. When a fistulous communica tion between the aorta and the lung is suspected, aorto graphy and bronchoscopy should be performed very care Fig. 4. The wall of the aneurysm (center) consists only of dense fully (7). In patients with hemoptysis, CT and MRI, fibrous tissue. The aneurysm is lined by thrombus (bottom) (HE stain, which provide considerable diagnostic information, should xlOO). first be performed, regardless of whether chest radiograph Hemoptysis is the most common, but not specific, shows anyofabnormal shadow. symptom aortobronchopulmonary fistula. Although Discussion fistula can occur not only secondary to operation but also as a result of pathological processes unrelated to prior Although false ascending aortic aneurysm following operation (5), history of cardiosurgery is a key factor in the cardiosurgery has been reported (1-5), it is rare (3). In consideration of the diagnosis of a fistula. Infections as a modern times, such aneurysms are usually the result of result of surgery have become more infrequent, and postoperative mediastinitis or septicemia, aortic graft postoperative fistulas do not always result from infections degeneration, or technical problems of surgical procedures (3-5, 9-ll). In a case such as the present case, in which (3-6). Most originate at the site of aortic cannulation or there was overt infection, however, it is necessary to con aortotomy suture (1-3, 5-7). The patient in the present sider the possibility of fatal false aneurysm. Even the case had suffered from prolonged and severe mediastinitis passage of considerable time between surgery and and osteomyelitis of the sternum. Infection may cause hemoptysis or chronic minor hemorrhage does not preclude weakness of the aortic wall at the site of aortotomy suture the possibility of fistula (1-5). Awareness, early recogni and subsequent formation of false aneurysm. The aneurysm tion and surgical repair of fistulas will lower their high invades the lung, by either pressure necrosis or mortality rates (8). References Hemoptysis due topresumably aortobronchopulmonary fistula is chronic inflammation, and manifests as hemoptysis. usually fatal if untreated (1, 2, 4). Therefore, more than 1) Coblentz CL, Sallee DS, Chiles C. Aortobronchopulmonary fistula half of all diagnoses are made at autopsy (8). The fact that complicating aortic aneurysm: diagnosis in four cases. Am J Roentg the fistula in the present case was very small and might 150: 535, 1988. have been obstructed by a clot in the aneurysm may have 2) Wheeler AP, Loyd JE. Fatal hemoptysis: aortobronchial fistula as prevented fatal massive hemoptysis. Because hemoptysis in a preventable cause of death. Crit Care Med 17: 1228, 1989. 3) Soorae AS, Cleland J, O'Kane H. Delayed nonmycotic false this patient recurred over the course of one year, there was aneurysm of ascending aorta cannulation site. Thorax 32: 743, 1977. sufficient time to perform the several examinations necessary 4) Oldham KT, Johnsen K, Winterscheid L, Larson EB. Remembrance to precisely diagnose its source and to administer effective of things past: aortobronchial fistula 15 years after thoracic aortic homograft. West J Med 139: 225, 1983. treatment. Chest radiograph provided little diagnostic in 5) Graeber GM, Farrell BG Jr, Neville JF Jr, Parker FB. Successful formation (8). Furthermore, minor hemoptysis does not diagnosis and management of fistulas between the aorta and the indicate the presence of a fistula which enlarges and tracheobronchial tree. Ann Thorac Surg 29: 555, 1980. precipitates lethal hemorrhage. CT and MRI clearly revealed 6) Garrett HE, Ricks RK, Lewis JM, Howell JF, DeBakey ME. the existence of a saccular shadow adjacent to the ascending Hemoptysis secondary to aortopulmonary fistula. A report of two cases of successful treatment by operation. J Thorac Cardiovasc aorta as well as its communication with the aortic lumen, Surg 49: 588, 1963. 7) Ramakantan R, Shah P. False aneurysm secondary to aortic confirming that these noninvasive, easy and safe methods cannulation-rupture into lung with fatal hemoptysis during aortoof examination are very useful in the diagnosis of graphy. Thorac Cardiovasc Surg 37: 322, 1989. 8) Demeter SL, Cordasco EM. Aortobronchial fistula: keys to aneurysms. The fistula, however, was not demonstrable by successful management. Angiology 31: 431, 1980. CT, MRI or aortography, rather it was found intra operatively. Fatal rupture of a false aneurysm during 1045 Internal Medicine using Vol. an 31, injection No. 8 (August 1992) i.e., at high aortography machine,
Ishidaetal 9) Davey MG. Aorto-pulmonary fistula due to failure of an Ivalon aorta. Ann Thorac Surg 2: 82, 1966. graft for coarctation of the aorta. Thorax 17: 363, 1962. ll) Symbas PN, Hunter RM, Vlasis SE, Ansley JD. Infected descending 10) Neville WE, Correll NO Jr, Maben H. Resection of aortic-bronchial aortic fistula. Ann Thorac Surg 41: 647, 1986. fistula occurring ten years after graft replacement of the descending
1046
Internal
Medicine
Vol. 31, No. 8 (August
1992)
Intro duction
negative, and tuberculin skin test was doubtful. Chest radiograph showed granular and reticular shadows at False aneurysm is a rare complication of aortotomy per bilateral lower lung fields and a questionable contour formed during cardiosurgery. Although hemoptysis due to adjacent to the right upper cardiac silhouette (Fig. 1). Bronchoscopy revealed no source of hemoptysis, and aortobronchopulmonary fistula of the aneurysm is usually bronchography revealed no specific findings for bron fatal if untreated, recurrent hemoptysis as the sole symp chiectasis. CT showed a 4 x 4 cm saccular shadow anterior tom is most unusual. We report such a case in which com and to the right of the ascending aorta. This shadow was puted tomography (CT) and magnetic resonance imaging partially enhanced to the same intensity as the aortic lumen, (MRI) were very useful in the diagnosis of the aneurysm suggesting a saccular aneurysm many clots (Fig. 2A). and in which surgery was successful. MRI also clearly showed that thewith shadow had a communica Case Report tion with the lumen of the ascending aorta (Fig. 2B). Aortography at the root of the aorta revealed that a small A 54-year-old man was referred to our hospital for re quantity of contrast medium had leaked out of the current hemoptysis of one year duration. The patient had ascending aorta, and insertion of the catheter into the undergone repair of rupture of the sinus of Valsalva aneurysm was then attempted. Although manual injection of a small amount of contrast medium into the aneurysm sustained inhea had traffic accident six years prior to admission. Thereafter, suffered mediastinitis and osteomyelitis confirmed that this shadow was a false aneurysm arising of the sternum, and the rectus abdominal muscle had been from the ascending aorta, no aortobronchopulmonary transplanted in a sternotomy chest wound. The patient was asymptomatic, and neither heart Intraoperatively, aneurysm found communicationsthe were detectedwas (Fig. 3). to be mostly filled with thrombotic material, and a 10 mm diameter rent murmurs nor abnormal breathing sounds were noted. Pulse in the aorta on the superior surface of the site of previous was regular at 66/min, and blood pressure was 140/86 aortotomy suture was visible. Removal of the thrombi mmHg. Hemoglobin concentration was 13.4 g/dl, and disclosed a 1 mm diameter fistula on the right floor of the leukocyte count was 5.9 x lOVmm3. C-reactive protein was aneurysm. Artificial ventilation demonstrated leakage of air negative. Examination of the sputum showed no malignant into the aneurysm via the fistula, confirming its comcells or tubercle bacilli. Blood serology for syphilis was From the Department of Internal Medicine and *the Department of Cardiovascular Surgery, Tosei General Hospital, Seto Received for publication April 17, 1991; Accepted for publication May 6, 1992 Reprint requests should be addressed to Dr. Akihiro Ishida, the Department of Internal Medicine, Tosei General Hospital, 160 Nishioiwak Seto489, Japan Internal
Medicine
Vol. 31, No. 8 (August
1992)
1043
Ishida
et al munication with the lung. Histologic examination showed that the aneurysm was of the false variety (Fig. 4). The patient's postoperative course was uneventful.
Fig. 1. Chest radiograph shows a questionable contour adjacent Fig. 3. Injection of contrast medium into the aneurysm reveals it to the right upper cardiac silhouette (arrow). to be a false aneurysm arising from the ascending aorta.
Fig. 2. A) CT at the level of bifurcation of pulmonary artery shows a saccular shadow filled with thrombus (left arrow) with enhance ment equaling that of the aortic lumen (right arrow). B) MRI shows this shadow to have a communication with the aortic lumen (arrow 1 044
Internal
Medicine
Vol. 31,
No. 8 (August
1992)
Hemoptysis from Postoperative
Aneurysm
pressure, in a patient with chronic hemoptysis has been reported (7). Fortunately, in the present case, low pressure manual injection of contrast medium into the aneurysm caused no massive hemoptysis as a result of the dislodge ment of the clot obstructing the fistula. Injection of even a small amount of contrast medium provided satisfactory images. Frequently, aortography is essential to formulate the decisions regarding surgical procedures. Bronchoscopy is helpful only in the diagnosis of an endobronchial tumor as the source of the bleeding and, for the same reason as in aortography, there is a risk of lethal hemorr hage associated with its use. When a fistulous communica tion between the aorta and the lung is suspected, aorto graphy and bronchoscopy should be performed very care Fig. 4. The wall of the aneurysm (center) consists only of dense fully (7). In patients with hemoptysis, CT and MRI, fibrous tissue. The aneurysm is lined by thrombus (bottom) (HE stain, which provide considerable diagnostic information, should xlOO). first be performed, regardless of whether chest radiograph Hemoptysis is the most common, but not specific, shows anyofabnormal shadow. symptom aortobronchopulmonary fistula. Although Discussion fistula can occur not only secondary to operation but also as a result of pathological processes unrelated to prior Although false ascending aortic aneurysm following operation (5), history of cardiosurgery is a key factor in the cardiosurgery has been reported (1-5), it is rare (3). In consideration of the diagnosis of a fistula. Infections as a modern times, such aneurysms are usually the result of result of surgery have become more infrequent, and postoperative mediastinitis or septicemia, aortic graft postoperative fistulas do not always result from infections degeneration, or technical problems of surgical procedures (3-5, 9-ll). In a case such as the present case, in which (3-6). Most originate at the site of aortic cannulation or there was overt infection, however, it is necessary to con aortotomy suture (1-3, 5-7). The patient in the present sider the possibility of fatal false aneurysm. Even the case had suffered from prolonged and severe mediastinitis passage of considerable time between surgery and and osteomyelitis of the sternum. Infection may cause hemoptysis or chronic minor hemorrhage does not preclude weakness of the aortic wall at the site of aortotomy suture the possibility of fistula (1-5). Awareness, early recogni and subsequent formation of false aneurysm. The aneurysm tion and surgical repair of fistulas will lower their high invades the lung, by either pressure necrosis or mortality rates (8). References Hemoptysis due topresumably aortobronchopulmonary fistula is chronic inflammation, and manifests as hemoptysis. usually fatal if untreated (1, 2, 4). Therefore, more than 1) Coblentz CL, Sallee DS, Chiles C. Aortobronchopulmonary fistula half of all diagnoses are made at autopsy (8). The fact that complicating aortic aneurysm: diagnosis in four cases. Am J Roentg the fistula in the present case was very small and might 150: 535, 1988. have been obstructed by a clot in the aneurysm may have 2) Wheeler AP, Loyd JE. Fatal hemoptysis: aortobronchial fistula as prevented fatal massive hemoptysis. Because hemoptysis in a preventable cause of death. Crit Care Med 17: 1228, 1989. 3) Soorae AS, Cleland J, O'Kane H. Delayed nonmycotic false this patient recurred over the course of one year, there was aneurysm of ascending aorta cannulation site. Thorax 32: 743, 1977. sufficient time to perform the several examinations necessary 4) Oldham KT, Johnsen K, Winterscheid L, Larson EB. Remembrance to precisely diagnose its source and to administer effective of things past: aortobronchial fistula 15 years after thoracic aortic homograft. West J Med 139: 225, 1983. treatment. Chest radiograph provided little diagnostic in 5) Graeber GM, Farrell BG Jr, Neville JF Jr, Parker FB. Successful formation (8). Furthermore, minor hemoptysis does not diagnosis and management of fistulas between the aorta and the indicate the presence of a fistula which enlarges and tracheobronchial tree. Ann Thorac Surg 29: 555, 1980. precipitates lethal hemorrhage. CT and MRI clearly revealed 6) Garrett HE, Ricks RK, Lewis JM, Howell JF, DeBakey ME. the existence of a saccular shadow adjacent to the ascending Hemoptysis secondary to aortopulmonary fistula. A report of two cases of successful treatment by operation. J Thorac Cardiovasc aorta as well as its communication with the aortic lumen, Surg 49: 588, 1963. 7) Ramakantan R, Shah P. False aneurysm secondary to aortic confirming that these noninvasive, easy and safe methods cannulation-rupture into lung with fatal hemoptysis during aortoof examination are very useful in the diagnosis of graphy. Thorac Cardiovasc Surg 37: 322, 1989. 8) Demeter SL, Cordasco EM. Aortobronchial fistula: keys to aneurysms. The fistula, however, was not demonstrable by successful management. Angiology 31: 431, 1980. CT, MRI or aortography, rather it was found intra operatively. Fatal rupture of a false aneurysm during 1045 Internal Medicine using Vol. an 31, injection No. 8 (August 1992) i.e., at high aortography machine,
Ishidaetal 9) Davey MG. Aorto-pulmonary fistula due to failure of an Ivalon aorta. Ann Thorac Surg 2: 82, 1966. graft for coarctation of the aorta. Thorax 17: 363, 1962. ll) Symbas PN, Hunter RM, Vlasis SE, Ansley JD. Infected descending 10) Neville WE, Correll NO Jr, Maben H. Resection of aortic-bronchial aortic fistula. Ann Thorac Surg 41: 647, 1986. fistula occurring ten years after graft replacement of the descending
1046
Internal
Medicine
Vol. 31, No. 8 (August
1992)
