A Patient with Aortic Arch Aneurysm Perforating the Left Pulmonary Artery Daisuke Yuji, Ikuo Katayama, and Masashi Tanaka, Japan
An aortic arch aneurysm rarely perforates the pulmonary artery, but once this occurs symptoms of heart failure may develop rapidly and result in a serious course. Here, we report such a case that was treated with life-saving emergency surgery. The patient was an 86-year-old man in whom aortic arch aneurysm had been pointed out 8 years earlier, but left untreated. In January 2014, dyspnea developed and he visited the emergency unit of our hospital. Continuous murmur was heard on auscultation, and aorta-pulmonary artery shunt was noted on transthoracic echocardiography. Chest computed tomography revealed a giant aortic arch aneurysm of size 106 mm that had perforated the left pulmonary artery (LPA). Emergency surgery was performed for a diagnosis of acute heart failure associated with perforation of the LPA by a giant aortic arch aneurysm. The postoperative course was favorable, and the patient was discharged with independent walking on postoperative day 28.
Perforation of the pulmonary artery by an aortic arch aneurysm is relatively rare. Boyd et al.1 found the disease in 45 (3.7%) of 1,197 autopsy cases of thoracic aortic aneurysm rupture. The first successful surgery for this disease was described in 1960 by Giacobine and Cooley,2 but has only occasionally been reported thereafter. Once the condition occurs, heart failure develops rapidly because of left-to-right shunt from the aorta to the pulmonary artery and life-saving surgery may be difficult. Here, we report a case of perforation of the pulmonary artery by an aortic arch aneurysm that was treated successfully by emergency surgery.
CASE REPORT The patient was an 86-year-old man with a chief complaint of dyspnea. His medical history included abdominal aortic Conflict of Interest: None of the authors have a conflict of interest. Department of Cardiovascular Surgery, Shonan Fujisawa Tokushukai Hospital, Fujisawa city Kanagawa, Japan. Correspondence to: Daisuke Yuji, MD, Department of Cardiovascular Surgery, Shonan Fujisawa Tokushukai Hospital, Japan; E-mail: [email protected] Ann Vasc Surg 2015; 29: 1658.e1–1658.e4 http://dx.doi.org/10.1016/j.avsg.2015.05.027 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: October 25, 2014; manuscript accepted: May 27, 2015; published online: July 16, 2015.
replacement 8 years earlier and hypertension. He had no relevant familial medical history. Thoracic aortic aneurysm had been pointed out 8 years ago when he underwent surgery for abdominal aortic aneurysm, but had been left untreated. Hoarseness developed about 1 year ago. In January 2014, he suddenly developed dyspnea during sleep and this did not remit. Thus, he visited the emergency outpatient unit of our hospital. At admission, height was 166 cm; body weight 56 kg; consciousness, clear; blood pressure, 120/62 mm Hg; pulse, 110/min, irregular; SPO2, 94% (6 L); and respiratory rate, 24/min with dyspnea. Levine III/VI continuous murmur with the strongest sound in 3 left sternal border was heard on chest auscultation. Edema was noted in the bilateral crura. Laboratory tests at admission gave arterial blood gas (O2 60% mask) pH 7.388, pCO2 38.4 mm Hg, pO2 79.8 mm Hg, base excess 1.6 mmol/L, HCO3 22.6 mmol/L, white blood cell 8,000/mL, red blood cell 346/mL, hemoglobin 10.4 g/dL, platelet 10.2/mL, C-reactive protein 3.43 mg/dL, blood urea nitrogen 29.4 mg/dL, createnine 1.52 mg/dL, brain natriuretic peptide 495 pg/mL; and D-dimer 104 mg/mL. In chest radiography (Fig. 1), a shadow of the aortic arch aneurysm was noted in the upper left lung field. The trachea was excluded rightward, and congestion was noted in the bilateral lung fields. cardio thoracic ratio was 65.8%. Transthoracic echocardiography (Fig. 2) gave values of left ventricular end-diastolic dimension 46 mm, left ventricular internal dimension in systole 28 mm, and ejection fraction 69%. Left ventricular wall movement was normal. The right side of the heart was slightly dilated. Shunt flow from the aortic arch to the left pulmonary artery (LPA) was noted. Chest contrast computed
1658.e1
1658.e2 Case reports
Fig. 1. X-ray showed cardiomegaly, pulmonary congestion, and enlargement of aortic aorta. tomography (CT; Fig. 3) revealed a giant aortic arch aneurysm with a maximum minor axis of 106 mm, and the LPA was partially perforated in the sagittal view. Based on these findings, the patient was diagnosed with acute heart failure caused by perforation of the LPA by the aortic arch aneurysm and underwent immediate emergency surgery. The target region was approached by median sternotomy. An 8-mm blood supply tube was inserted into the ascending aorta, and an 18F blood supply tube for debris removal was inserted into the right femoral artery. A 2-stage blood removal tube was inserted through the right atrium, followed by insertion of an left atrium-left ventricule vent to establish extracorporeal circulation. The root vent was inserted into the ascending aorta, and cooling of the whole body was initiated. Thrill was palpated on the cranial side of the LPA. Pulmonary artery pressure rose and the shunt region were manually excluded to control inflow into the pulmonary artery. Circulation was arrested at a pharyngeal temperature of 28 C, and the aorta was incised. Catheters for selective cerebral perfusion were inserted into the 3 branches of the arch, and perfusion was initiated. Mural thrombus and atheroma in the aneurysm were removed as much as possible. The peripheral side of the anastomosis line was set, 13 horizontal mattress sutures were applied using 2-0 Teffdessor, and the vessel was anastomosed with a 26mm 4-branched J graft with an open distal knotted suture during circulatory arrest of the lower half of the body. Observation of the main trunk of the pulmonary artery after a longitudinal incision of about 30 mm showed the presence of a perforated region of about 10 mm in the ceiling near the bifurcation of the LPA (Fig. 4). The perforated region was closed with 2 pledgeted sutures with 4-0 polypropylene thread, and the incised region of the pulmonary artery was closed by continuous 2-layer suture with 4-0 polypropylene thread. Blood supply through the right femoral artery was restarted, debris was removed, and circulation was restarted with an
Annals of Vascular Surgery
antegrade blood supply through the lateral branch of the graft, followed by rewarming. Open proximal anastomosis was applied to the central side 5 mm above the sinotubular junction by continuous suture with 4-0 polypropylene thread. After removal of air from the true lumen, the blockage of the graft was released. Finally, the branches of the arch were individually reconstructed. Weaning from the artificial heart-lung apparatus was straightforward, and there was no hemorrhage from the pulmonary artery. The operative time was 340 min, including cardiopulmonary bypass of 117 min, selective cerebral perfusion of 114 min, and circulatory arrest of the lower half of the body of 90 min. The postoperative course was favorable. Tubes were removed on postoperative day (POD) 2, and the patient was discharged from the intensive care unit on POD 5. Thoracentesis was performed for pleural effusion, but the patient recovered and was discharged with independent walking on POD 28. The patient is being followed at the outpatient clinic, and the course has been favorable.
DISCUSSION Perforation of the pulmonary artery by an aortic arch aneurysm is a rare1 but life-threatening2 condition because of rapid development of heart failure due to shunt blood flow. Fistula formation may also occur between the aortic aneurysm and pulmonary arterial wall because of friction with the pulmonary artery as the aortic aneurysm expands and because of infection or inflammation in the region between the aneurysm and pulmonary artery.3 The condition may be caused by syphilitic infection,4 acute aortic dissection,5 trauma,6 complication in an ascending aortic aneurysm associated with Marfan syndrome,7 and aneurysm formation in the anastomosed region after aortic replacement.8 In addition to CT for diagnosis, SwaneGanz catheterization is useful to treat heart failure and measure shunt, and echocardiography is effective for visualizing the shunt as a low-invasive technique. Since shunt increases with time, rapid surgical treatment is required after diagnosis. The method for repair of the perforated region of the pulmonary artery includes closure by continuous suture and patching with autologous pericardium, equine pericardium, and (expanded polytetrafluorethylene) ePTFE. In our patient, the perforation was small and was closed by 2 pledgeted sutures with 4-0 polypropylene thread. To our knowledge, the first case of perforation of the pulmonary artery by a true aortic arch aneurysm in Japan was described by Oka et al.9 and a total of 15 cases have now been reported.8e22 In these cases, the male:female ratio was 10:5, the mean age was 72.4 ± 7.9 years old (55e82 years old), and the
Vol. 29, No. 8, November 2015
Case reports 1658.e3
Fig. 2. Echogram showed shunt flow (white arrow) from arch aneurysm into LPA.
Fig. 3. (A) Preoperative CT showed an aortic arch aneurysm with a maximum diameter of 106 mm. (B) Preoperative CT showed aortic aneurysm was communicated with pulmonary artery resulting in aortopulmonary fistula (black arrow).
most frequent chief complaint was dyspnea. Hoarseness, malaise, and chest pain were also noted. Thirteen cases were diagnosed by CT and SwaneGanz catheterization was applied in 10 cases. Diagnosis was also made by echocardiography in some cases, and one case was definitely diagnosed by aortography.9 Surgery was performed in 13 cases and one case was conservatively treated because of a request from the patient, although the disease was definitely diagnosed.10 One case was diagnosed at autopsy.11 The perforated region of the pulmonary artery was closed by suture in 6 cases9,12e16 and with a patch in 6 cases.8,13,17e20 The patch material was autologous pericardium in 2 cases,17,18 equine pericardium in 2 cases,8,13 ePTFE in 1 case,20 and not described in 1 case.19 In one case, closure by suture
and patch was difficult because the pulmonary artery was dissected, and vascular replacement with ePTFE was performed.21 Ten patients recovered and were discharged, but 5 patients died: 1 patient who died 14 days after onset because of refusing surgery despite the disease being definitely diagnosed,10 1 was definitely diagnosed at autopsy,11 and 3 died after surgery.9,13,16 In one of these 3 patients, the disease was definitely diagnosed 3 days after onset, and emergency surgery was performed on the same day, but the patient died because of postoperative cerebral infarction and disseminated intravascular coagulation.9 The other 2 cases13,16 were diagnosed as acute aggravation of chronic heart failure in an outpatient examination and were initially treated for heart failure. The disease was definitely
1658.e4 Case reports
Fig. 4. Intraoperative view: aortopulmonary fistula (10 mm diameter) (black arrow) was observed, which closed with 4-0 polypropylene, with pledget.
diagnosed after aggravation of symptoms of heart failure and surgery was performed 9 days after onset, but postoperative disseminated intravascular coagulation and multiple organ failure developed and both patients died 34 days after onset. Since shunt increases with time in this disease, early diagnosis, treatment of heart failure, and surgery are important. As a physical finding, continuous murmur was heard on auscultation in many cases and this requires rapid investigation of the cause using chest CT, echocardiography, and SwaneGanz catheterization, followed by surgery in the early phase. In our patient, aortic arch aneurysm was suspected on chest X-ray photograph performed at the emergency outpatient unit, and perforation of the pulmonary artery by aortic arch aneurysm was confirmed as the cause of continuous murmur by echocardiography and chest contrast CT, which enabled rapid diagnosis and treatment of the cause of rapid heart failure. Rapid aggravation of heart failure after perforation of the pulmonary artery by an aortic arch aneurysm is rare, and lifesaving surgery is difficult if an accurate diagnosis is not made at an early stage. Therefore, despite its rarity, this disease should be considered and heart failure should be examined in patients with a thoracic aortic aneurysm. In conclusion, in the case reported here, perforation of the pulmonary artery by an aortic arch aneurysm manifested as acute heart failure. Treatment with aortic arch replacement and pulmonary artery repair achieved a favorable outcome. REFERENCES 1. Boyd LJ. A study of four thousand reported cases of aneurysm of the thoracic aorta. Am J Med Sci 1924;168:654e63.
Annals of Vascular Surgery
2. Giacobine JW, Cooley DA. Surgical treatment of aortopulmonary fistula secondary to aortic arch aneurysm: report of a successful case. J Thorac Cardiovasc Surg 1960;39: 130e6. 3. Pessotto R, Santini F, Bertolini P, et al. Surgical treatment of an aortopulmonary artery fistula complicating a syphilitic aortic aneurysm. Cardiovasc Surg 1995;3:707e10. 4. Adhyapak SM, Haridas AK, Yeriswamy MC, et al. Syphilitic aortitis; an uncommon cause of acquired aortopulmonary fistula. Ann Thorac Surg 2009;88:1672e4. 5. Piciche M, De Paulis R, Chiariello L. A review of aortopulmonary fistulas in aortic dissection. Ann Thorac Surg 1999;68:1833e6. 6. Dixit MD, Gan M, Narendra NG. Aortopulmonary fistula; rare complication of aortic aneurysm. Tex Heart Inst J 2009;36:483e5. 7. Messetti M, Babatasi G, Rossi A, et al. Aortopulmonary fistula; an uncommon complication in dystrophic aortic aneurysm. Ann Thorac Surg 1995;59:1563e4. 8. Suzuki K, Hamano K, Hanada S, et al. Successful surgical management of an aortic arch aneurysm with an aortopulmonary artery fistula. J Cardiovasc Surg 2003;32:137e40. 9. Oka Y, Fujita T, Nakajima N, et al. A surgical case report of aortic aneurysm with rupture into the pulmonary artery. J Thorac Cardiovasc Surg 1980;28:147e54. 10. Nara, I., Fujiwara, T., Kin, S., et al. Daidoumyaku-haidoumyakurou wo shoji usokunomi no haiukketsu to taryou no kyousui wo mitometa kyuubudaidoumyakuryuu no ichi rei: The medical journal of akita city hospital. 21 : 15e20, 2012. 11. Fujimori S, Sakai M, Iwao K, et al. Rupture of aortic aneurysm into pulmonary artery with occluded orifice by thrombus. Shinzou 1988;20:1007e13. 12. Kakiko T, Konishi T, Maruyama S, et al. A case of successful repair of aorto-pulmonary artery fistula secondary to aortic arch aneurysm. Kokyu To Junkan 1992;40:603e7. 13. Daidoji H, Nitobe J, Watanabe T, et al. A case of acute heart failure due to abrupt rupture of aortic aneurysm into pulmonary artery. J Cardiol 2011;90e2. 14. Shirai J, Imoto K, Uchida K, et al. A case report of aortopulmonary fistula caused by rupture of an aortic arch aneurysm. J Jpn Surg Assoc 2012;73:3086e90. 15. Jinnouchi K, Ohteki H, Naito K, et al. Successful surgical treatment of an arch aneurysm with pulmonary artery fistula. J Vasc Surg 2012;21:687e90. 16. Takahashi A, Uchida T, Kim C, et al. Aortopulmonary fistula caused by aortic arch aneurysm rupture presenting severe left to right shunt and congestive heart failure. Kyobu Geka 2013;66:532e6. 17. Kameyama T, Nakayama S, Okabayashi H, et al. A case report of successful repair of an aortopulmonary fistula with partial dissection of the pulmonary artery. J Thorac Cardiovasc Surg 1992;40:432e4. 18. Yoshida M, Mukohara N, Obo H, et al. A case of huge aortic arch aneurysm with unrevealed aortopulmonary fistula resulting intraoperative pulmonary artery rupture after total arch replacement. J Cardiovasc Surg 2004;33:403e6. 19. Kaki N, Irie K, Hata I, et al. Aortopulmonary fistula in aortic arch aneurysm treated successfully by surgery. J Vasc Surg 2005;14:105e7. 20. Iwahashi K, Cho C, Iwasaki T, et al. Aortic arch aneurysm with aortopulmonary fistula. Kyobu Geka 2007;60:853e6. 21. Nishimura Y, Okamura Y, Uchita S, et al. Abrupt rupture of an aortic arch aneurysm into pulmonary artery. Eur J Cardiothorac Surg 2009;36:212e3. 22. Okada T, Nakai M, Shimamoto M, et al. A case of aortopulmonary artery fistula. J Cardiovasc Surg 2012;41:195e9.
An aortic arch aneurysm rarely perforates the pulmonary artery, but once this occurs symptoms of heart failure may develop rapidly and result in a serious course. Here, we report such a case that was treated with life-saving emergency surgery. The patient was an 86-year-old man in whom aortic arch aneurysm had been pointed out 8 years earlier, but left untreated. In January 2014, dyspnea developed and he visited the emergency unit of our hospital. Continuous murmur was heard on auscultation, and aorta-pulmonary artery shunt was noted on transthoracic echocardiography. Chest computed tomography revealed a giant aortic arch aneurysm of size 106 mm that had perforated the left pulmonary artery (LPA). Emergency surgery was performed for a diagnosis of acute heart failure associated with perforation of the LPA by a giant aortic arch aneurysm. The postoperative course was favorable, and the patient was discharged with independent walking on postoperative day 28.
Perforation of the pulmonary artery by an aortic arch aneurysm is relatively rare. Boyd et al.1 found the disease in 45 (3.7%) of 1,197 autopsy cases of thoracic aortic aneurysm rupture. The first successful surgery for this disease was described in 1960 by Giacobine and Cooley,2 but has only occasionally been reported thereafter. Once the condition occurs, heart failure develops rapidly because of left-to-right shunt from the aorta to the pulmonary artery and life-saving surgery may be difficult. Here, we report a case of perforation of the pulmonary artery by an aortic arch aneurysm that was treated successfully by emergency surgery.
CASE REPORT The patient was an 86-year-old man with a chief complaint of dyspnea. His medical history included abdominal aortic Conflict of Interest: None of the authors have a conflict of interest. Department of Cardiovascular Surgery, Shonan Fujisawa Tokushukai Hospital, Fujisawa city Kanagawa, Japan. Correspondence to: Daisuke Yuji, MD, Department of Cardiovascular Surgery, Shonan Fujisawa Tokushukai Hospital, Japan; E-mail: [email protected] Ann Vasc Surg 2015; 29: 1658.e1–1658.e4 http://dx.doi.org/10.1016/j.avsg.2015.05.027 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: October 25, 2014; manuscript accepted: May 27, 2015; published online: July 16, 2015.
replacement 8 years earlier and hypertension. He had no relevant familial medical history. Thoracic aortic aneurysm had been pointed out 8 years ago when he underwent surgery for abdominal aortic aneurysm, but had been left untreated. Hoarseness developed about 1 year ago. In January 2014, he suddenly developed dyspnea during sleep and this did not remit. Thus, he visited the emergency outpatient unit of our hospital. At admission, height was 166 cm; body weight 56 kg; consciousness, clear; blood pressure, 120/62 mm Hg; pulse, 110/min, irregular; SPO2, 94% (6 L); and respiratory rate, 24/min with dyspnea. Levine III/VI continuous murmur with the strongest sound in 3 left sternal border was heard on chest auscultation. Edema was noted in the bilateral crura. Laboratory tests at admission gave arterial blood gas (O2 60% mask) pH 7.388, pCO2 38.4 mm Hg, pO2 79.8 mm Hg, base excess 1.6 mmol/L, HCO3 22.6 mmol/L, white blood cell 8,000/mL, red blood cell 346/mL, hemoglobin 10.4 g/dL, platelet 10.2/mL, C-reactive protein 3.43 mg/dL, blood urea nitrogen 29.4 mg/dL, createnine 1.52 mg/dL, brain natriuretic peptide 495 pg/mL; and D-dimer 104 mg/mL. In chest radiography (Fig. 1), a shadow of the aortic arch aneurysm was noted in the upper left lung field. The trachea was excluded rightward, and congestion was noted in the bilateral lung fields. cardio thoracic ratio was 65.8%. Transthoracic echocardiography (Fig. 2) gave values of left ventricular end-diastolic dimension 46 mm, left ventricular internal dimension in systole 28 mm, and ejection fraction 69%. Left ventricular wall movement was normal. The right side of the heart was slightly dilated. Shunt flow from the aortic arch to the left pulmonary artery (LPA) was noted. Chest contrast computed
1658.e1
1658.e2 Case reports
Fig. 1. X-ray showed cardiomegaly, pulmonary congestion, and enlargement of aortic aorta. tomography (CT; Fig. 3) revealed a giant aortic arch aneurysm with a maximum minor axis of 106 mm, and the LPA was partially perforated in the sagittal view. Based on these findings, the patient was diagnosed with acute heart failure caused by perforation of the LPA by the aortic arch aneurysm and underwent immediate emergency surgery. The target region was approached by median sternotomy. An 8-mm blood supply tube was inserted into the ascending aorta, and an 18F blood supply tube for debris removal was inserted into the right femoral artery. A 2-stage blood removal tube was inserted through the right atrium, followed by insertion of an left atrium-left ventricule vent to establish extracorporeal circulation. The root vent was inserted into the ascending aorta, and cooling of the whole body was initiated. Thrill was palpated on the cranial side of the LPA. Pulmonary artery pressure rose and the shunt region were manually excluded to control inflow into the pulmonary artery. Circulation was arrested at a pharyngeal temperature of 28 C, and the aorta was incised. Catheters for selective cerebral perfusion were inserted into the 3 branches of the arch, and perfusion was initiated. Mural thrombus and atheroma in the aneurysm were removed as much as possible. The peripheral side of the anastomosis line was set, 13 horizontal mattress sutures were applied using 2-0 Teffdessor, and the vessel was anastomosed with a 26mm 4-branched J graft with an open distal knotted suture during circulatory arrest of the lower half of the body. Observation of the main trunk of the pulmonary artery after a longitudinal incision of about 30 mm showed the presence of a perforated region of about 10 mm in the ceiling near the bifurcation of the LPA (Fig. 4). The perforated region was closed with 2 pledgeted sutures with 4-0 polypropylene thread, and the incised region of the pulmonary artery was closed by continuous 2-layer suture with 4-0 polypropylene thread. Blood supply through the right femoral artery was restarted, debris was removed, and circulation was restarted with an
Annals of Vascular Surgery
antegrade blood supply through the lateral branch of the graft, followed by rewarming. Open proximal anastomosis was applied to the central side 5 mm above the sinotubular junction by continuous suture with 4-0 polypropylene thread. After removal of air from the true lumen, the blockage of the graft was released. Finally, the branches of the arch were individually reconstructed. Weaning from the artificial heart-lung apparatus was straightforward, and there was no hemorrhage from the pulmonary artery. The operative time was 340 min, including cardiopulmonary bypass of 117 min, selective cerebral perfusion of 114 min, and circulatory arrest of the lower half of the body of 90 min. The postoperative course was favorable. Tubes were removed on postoperative day (POD) 2, and the patient was discharged from the intensive care unit on POD 5. Thoracentesis was performed for pleural effusion, but the patient recovered and was discharged with independent walking on POD 28. The patient is being followed at the outpatient clinic, and the course has been favorable.
DISCUSSION Perforation of the pulmonary artery by an aortic arch aneurysm is a rare1 but life-threatening2 condition because of rapid development of heart failure due to shunt blood flow. Fistula formation may also occur between the aortic aneurysm and pulmonary arterial wall because of friction with the pulmonary artery as the aortic aneurysm expands and because of infection or inflammation in the region between the aneurysm and pulmonary artery.3 The condition may be caused by syphilitic infection,4 acute aortic dissection,5 trauma,6 complication in an ascending aortic aneurysm associated with Marfan syndrome,7 and aneurysm formation in the anastomosed region after aortic replacement.8 In addition to CT for diagnosis, SwaneGanz catheterization is useful to treat heart failure and measure shunt, and echocardiography is effective for visualizing the shunt as a low-invasive technique. Since shunt increases with time, rapid surgical treatment is required after diagnosis. The method for repair of the perforated region of the pulmonary artery includes closure by continuous suture and patching with autologous pericardium, equine pericardium, and (expanded polytetrafluorethylene) ePTFE. In our patient, the perforation was small and was closed by 2 pledgeted sutures with 4-0 polypropylene thread. To our knowledge, the first case of perforation of the pulmonary artery by a true aortic arch aneurysm in Japan was described by Oka et al.9 and a total of 15 cases have now been reported.8e22 In these cases, the male:female ratio was 10:5, the mean age was 72.4 ± 7.9 years old (55e82 years old), and the
Vol. 29, No. 8, November 2015
Case reports 1658.e3
Fig. 2. Echogram showed shunt flow (white arrow) from arch aneurysm into LPA.
Fig. 3. (A) Preoperative CT showed an aortic arch aneurysm with a maximum diameter of 106 mm. (B) Preoperative CT showed aortic aneurysm was communicated with pulmonary artery resulting in aortopulmonary fistula (black arrow).
most frequent chief complaint was dyspnea. Hoarseness, malaise, and chest pain were also noted. Thirteen cases were diagnosed by CT and SwaneGanz catheterization was applied in 10 cases. Diagnosis was also made by echocardiography in some cases, and one case was definitely diagnosed by aortography.9 Surgery was performed in 13 cases and one case was conservatively treated because of a request from the patient, although the disease was definitely diagnosed.10 One case was diagnosed at autopsy.11 The perforated region of the pulmonary artery was closed by suture in 6 cases9,12e16 and with a patch in 6 cases.8,13,17e20 The patch material was autologous pericardium in 2 cases,17,18 equine pericardium in 2 cases,8,13 ePTFE in 1 case,20 and not described in 1 case.19 In one case, closure by suture
and patch was difficult because the pulmonary artery was dissected, and vascular replacement with ePTFE was performed.21 Ten patients recovered and were discharged, but 5 patients died: 1 patient who died 14 days after onset because of refusing surgery despite the disease being definitely diagnosed,10 1 was definitely diagnosed at autopsy,11 and 3 died after surgery.9,13,16 In one of these 3 patients, the disease was definitely diagnosed 3 days after onset, and emergency surgery was performed on the same day, but the patient died because of postoperative cerebral infarction and disseminated intravascular coagulation.9 The other 2 cases13,16 were diagnosed as acute aggravation of chronic heart failure in an outpatient examination and were initially treated for heart failure. The disease was definitely
1658.e4 Case reports
Fig. 4. Intraoperative view: aortopulmonary fistula (10 mm diameter) (black arrow) was observed, which closed with 4-0 polypropylene, with pledget.
diagnosed after aggravation of symptoms of heart failure and surgery was performed 9 days after onset, but postoperative disseminated intravascular coagulation and multiple organ failure developed and both patients died 34 days after onset. Since shunt increases with time in this disease, early diagnosis, treatment of heart failure, and surgery are important. As a physical finding, continuous murmur was heard on auscultation in many cases and this requires rapid investigation of the cause using chest CT, echocardiography, and SwaneGanz catheterization, followed by surgery in the early phase. In our patient, aortic arch aneurysm was suspected on chest X-ray photograph performed at the emergency outpatient unit, and perforation of the pulmonary artery by aortic arch aneurysm was confirmed as the cause of continuous murmur by echocardiography and chest contrast CT, which enabled rapid diagnosis and treatment of the cause of rapid heart failure. Rapid aggravation of heart failure after perforation of the pulmonary artery by an aortic arch aneurysm is rare, and lifesaving surgery is difficult if an accurate diagnosis is not made at an early stage. Therefore, despite its rarity, this disease should be considered and heart failure should be examined in patients with a thoracic aortic aneurysm. In conclusion, in the case reported here, perforation of the pulmonary artery by an aortic arch aneurysm manifested as acute heart failure. Treatment with aortic arch replacement and pulmonary artery repair achieved a favorable outcome. REFERENCES 1. Boyd LJ. A study of four thousand reported cases of aneurysm of the thoracic aorta. Am J Med Sci 1924;168:654e63.
Annals of Vascular Surgery
2. Giacobine JW, Cooley DA. Surgical treatment of aortopulmonary fistula secondary to aortic arch aneurysm: report of a successful case. J Thorac Cardiovasc Surg 1960;39: 130e6. 3. Pessotto R, Santini F, Bertolini P, et al. Surgical treatment of an aortopulmonary artery fistula complicating a syphilitic aortic aneurysm. Cardiovasc Surg 1995;3:707e10. 4. Adhyapak SM, Haridas AK, Yeriswamy MC, et al. Syphilitic aortitis; an uncommon cause of acquired aortopulmonary fistula. Ann Thorac Surg 2009;88:1672e4. 5. Piciche M, De Paulis R, Chiariello L. A review of aortopulmonary fistulas in aortic dissection. Ann Thorac Surg 1999;68:1833e6. 6. Dixit MD, Gan M, Narendra NG. Aortopulmonary fistula; rare complication of aortic aneurysm. Tex Heart Inst J 2009;36:483e5. 7. Messetti M, Babatasi G, Rossi A, et al. Aortopulmonary fistula; an uncommon complication in dystrophic aortic aneurysm. Ann Thorac Surg 1995;59:1563e4. 8. Suzuki K, Hamano K, Hanada S, et al. Successful surgical management of an aortic arch aneurysm with an aortopulmonary artery fistula. J Cardiovasc Surg 2003;32:137e40. 9. Oka Y, Fujita T, Nakajima N, et al. A surgical case report of aortic aneurysm with rupture into the pulmonary artery. J Thorac Cardiovasc Surg 1980;28:147e54. 10. Nara, I., Fujiwara, T., Kin, S., et al. Daidoumyaku-haidoumyakurou wo shoji usokunomi no haiukketsu to taryou no kyousui wo mitometa kyuubudaidoumyakuryuu no ichi rei: The medical journal of akita city hospital. 21 : 15e20, 2012. 11. Fujimori S, Sakai M, Iwao K, et al. Rupture of aortic aneurysm into pulmonary artery with occluded orifice by thrombus. Shinzou 1988;20:1007e13. 12. Kakiko T, Konishi T, Maruyama S, et al. A case of successful repair of aorto-pulmonary artery fistula secondary to aortic arch aneurysm. Kokyu To Junkan 1992;40:603e7. 13. Daidoji H, Nitobe J, Watanabe T, et al. A case of acute heart failure due to abrupt rupture of aortic aneurysm into pulmonary artery. J Cardiol 2011;90e2. 14. Shirai J, Imoto K, Uchida K, et al. A case report of aortopulmonary fistula caused by rupture of an aortic arch aneurysm. J Jpn Surg Assoc 2012;73:3086e90. 15. Jinnouchi K, Ohteki H, Naito K, et al. Successful surgical treatment of an arch aneurysm with pulmonary artery fistula. J Vasc Surg 2012;21:687e90. 16. Takahashi A, Uchida T, Kim C, et al. Aortopulmonary fistula caused by aortic arch aneurysm rupture presenting severe left to right shunt and congestive heart failure. Kyobu Geka 2013;66:532e6. 17. Kameyama T, Nakayama S, Okabayashi H, et al. A case report of successful repair of an aortopulmonary fistula with partial dissection of the pulmonary artery. J Thorac Cardiovasc Surg 1992;40:432e4. 18. Yoshida M, Mukohara N, Obo H, et al. A case of huge aortic arch aneurysm with unrevealed aortopulmonary fistula resulting intraoperative pulmonary artery rupture after total arch replacement. J Cardiovasc Surg 2004;33:403e6. 19. Kaki N, Irie K, Hata I, et al. Aortopulmonary fistula in aortic arch aneurysm treated successfully by surgery. J Vasc Surg 2005;14:105e7. 20. Iwahashi K, Cho C, Iwasaki T, et al. Aortic arch aneurysm with aortopulmonary fistula. Kyobu Geka 2007;60:853e6. 21. Nishimura Y, Okamura Y, Uchita S, et al. Abrupt rupture of an aortic arch aneurysm into pulmonary artery. Eur J Cardiothorac Surg 2009;36:212e3. 22. Okada T, Nakai M, Shimamoto M, et al. A case of aortopulmonary artery fistula. J Cardiovasc Surg 2012;41:195e9.
