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Late Rupture of a Totally Thrombosed Abdominal Aortic Aneurysm: A Case Report and Literature Review
Late Rupture of a Totally Thrombosed Abdominal Aortic Aneurysm: A Case Report and Literature Review Christiana Anastasiadou, Sotirios Giannakakis, Anastasios Papapetrou, George Galyfos, Gerasimos Papacharalampous, and Chrisostomos Maltezos, Athens, Greece
Chronic totally thrombosed abdominal aortic aneurysms (AAAs) comprise a rare medical situation, with only a few cases reported in literature. Optimal management has been controversial, although an early risk for rupture is present. Therefore, we present a rare case of late rupture in a patient with a totally thrombosed AAA, and we discuss proper treatment.
Spontaneous thrombosis of an abdominal aortic aneurysm (AAA) is a rare condition, with totally thrombosed AAAs being associated with a continuous risk for rupture according to some authors.1 Although all the reports in literature refer to an aneurysm rupture within the first year after presentation, no case of late rupture has been reported to date.2 Therefore, we are presenting a rare case of late rupture in a patient with total AAA thrombosis treated originally with axillobifemoral bypass.
CASE REPORT A 69-year-old male patient was referred to our emergency department with a diagnosis of a ruptured AAA. His medical history included: arterial hypertension (not adequately controlled with medical treatment), coronary artery disease treated with coronary artery bypass grafting almost 6 years ago (1 year before the diagnosis of the AAA), chronic heart failure (ejection fracture ¼ 30%), cerebrovascular disease, right-sided internal carotid artery occlusion (under dual antiplatelet treatment), left-sided internal carotid endarterectomy, and tobacco and alcohol
Department of Vascular Surgery, KAT General Hospital, Athens, Greece. Correspondence to: George Galyfos, Department of Vascular Surgery, KAT General Hospital, 2 Nikis Street, Kifisia, 14561, Athens, Greece; E-mail: [email protected] Ann Vasc Surg 2018; 46: 368.e5–368.e8 http://dx.doi.org/10.1016/j.avsg.2017.07.043 Ó 2017 Elsevier Inc. All rights reserved. Manuscript received: June 29, 2017; manuscript accepted: July 25, 2017; published online: 6 September 2017
abuse. Additionally, he had a history of a spontaneously thrombosed AAA (including the common iliac arteries) that presented 5 years ago (original diameter: 5.8 cm; Fig. 1) with symptoms of intermittent claudication. Owing to the severity of the general condition, he had undergone an axillobifemoral bypass in another institution which resolved the symptoms of the peripheral ischemia. However, the patient was lost on follow-up. On admission, the patient was suffering from severe abdominal and back pain worsening for the last 4 days; he was hemodynamically stable, and a pulsatile abdominal mass was palpated during physical examination. The axillobifemoral bypass was patent with palpable peripheral pulses. Regarding diagnostic imaging, the patient underwent a computed tomography angiography (CTA) that revealed a ruptured infrarenal AAA (maximum diameter ¼ 7 cm) and confirmed the patency of the axillobifemoral prosthetic graft (Fig. 2). Considering treatment, the patient underwent an emergency laparotomy. After dissection of the retroperitoneal hematoma, we proceeded to division of the left renal vein, exposure and clamping of the infrarenal aortic neck, excision of the aortic sac, and evacuation of the intraluminal thrombus, and finally, we primarily ligated the aortic neck and bifurcation (double layer of sutures). The distal aorta and aortic bifurcation were occluded, and therefore, no in situ reconstruction was considered an option. During declamping, a hemorrhage of the right renal artery orifice presented due to aortic clamp injury and excessive calcification of the aorta. After suprarenal aortic clamping, the deficit of the arterial wall was sutured successfully and aortic clamping was removed, with blood supply to the right renal artery being preserved. Finally, a flap of greater omentum was sutured on the aortic stump for further protection.
368.e5
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Annals of Vascular Surgery
Fig. 1. Computed tomography angiography (CTA) showing a totally thrombosed abdominal aortic aneurysm (AAA, arrow) presented 5 years ago.
Fig. 2. Computed tomography angiography (CTA) showing a rupture of the aneurysmal sac (thick arrow) and the patent axillobifemoral bypass (thin arrow).
Postoperatively, the patient was transferred to intensive care unit for further treatment. Postoperative medical treatment included low-molecular-weight heparin prophylaxis, antiplatelet therapy, and intravenous
antibiotics (standard prophylaxis for 3 days). Owing to a persistent diarrheal syndrome (even after discontinuation of the antibiotics), the patient was discharged 25 days after the procedure. His course remains uneventful
Volume 46, January 2018
Case reports 368.e7
Fig. 3. Postoperative computed tomography angiography (CTA) showing the intact aortic stump just below the level of the renal arteries (thick arrow) and the patent axillobifemoral bypass (thin arrow).
almost 2 months after discharge. Postoperative imaging with CTA shows the intact aortic stump just below the level of the renal arteries and patency of the axillobifemoral bypass (Fig. 3).
DISCUSSION Chronic total AAA occlusion is a relatively rare condition, with reported incidence reaching 0.6e2.8%.1,2 Factors that are associated with AAA thrombosis and occlusion include thromboembolic disease, hypercoagulation states, neoplasm, trauma, fever, iliac artery occlusive disease, and states of low flow such as chronic heart failure.3,4 Clinical presentation in chronic cases most commonly includes intermittent claudication. However, acute thrombosis usually presents with acute lower extremities ischemia and/or renal-visceral ischemia or paraplegia, and it is associated with a high mortality risk (almost 50%).2e5 Regarding pathophysiology, experimental studies have concluded that the lack of blood flow within the aneurysm sac does not reduce the pulse pressure of the thrombus to the aneurysmatic wall, and therefore, the risk of rupture remains.6 Takagi et al.7 have compared systematic and intrathrombotic pressure at 3 points within a thrombosed AAA, and they found only a 1e5% pressure decrease of mean intrathrombotic pressure. Moreover, Filis et al.8 have also compared systematic and intrathrombotic pressures within an occluded AAA, and they have found no significant differences. Additionally, rupture could occur in case a thrombosed aortic sac continued to be pressurized
by collateral branches.6,9 However, in our case, the rupture presented later, and this could be probably attributed to partial depressurization of the sac by the axillobifemoral bypass. Finally, all the reports in literature refer to rupture within 1 year after presentation, with a rupture risk reaching up to 15% in 6 months.2,9 Although some authors have advocated that thrombus could protect from rupture by reducing the wall stress, others have suggested that thrombus could cause sac expansion.1 Thrombosis is considered to involve the interaction between the process of inflammation and coagulation that are deteriorated in patients with AAA.10 Furthermore, intraluminal thrombus has been shown to alter the normal pattern of oxygen supply within the aortic wall leading to local hypoxia, wall weakening, and increasing the risk for rupture.8,11 Compared to the thrombusfree wall, the wall covered by thrombus is characterized by apoptotic smooth muscle cells, fragmented elastin fibers, and inflammatory cells.1,8 Therefore, the thrombus itself could affect the structural integrity and stability of the wall, predisposing to rupture. Considering treatment, many authors have selected the conservative management although this has led to rupture in several cases.12 Endovascular repair of a totally thrombosed AAA would be prohibiting as well, and therefore, there are only limited reports in literature.13 Many other strategies have been described in the past such as thrombectomy, aneursymectomy, and aortoiliac/aortofemoral bypass grafting with promising results, although only small case series have been reported in literature as well.14 However, in high-risk
368.e8 Case reports
patients, such as our patient, axillobifemoral bypass would be an acceptable alternative without treating the aneurysm or conducting a laparotomy.15 In the past, some authors have suggested causing thrombosis to a partially thrombosed AAA with simultaneous axillobifemoral bypass, without reporting any late complication associated with the AAA itself.16 However, follow-up reported after such procedures is of limited duration, and no long-term results have been published. Additionally, preoperative evaluation of the patient regarding the original procedure and the decision not to treat the thrombosed AAA definitely is an issue for consideration. Finally, given the chronic occlusion of the aortic bifurcation and the high surgical risk of the patient, no in situ reconstruction was attempted. Furthermore, the ligated aortic stump was covered with omentum for reinforcement.
CONCLUSIONS Chronically thrombosed AAAs are associated with a significant risk for rupture, even in the long term. Therefore, even if such patients have undergone bypass surgery or if they are under anticoagulant treatment, they should be under close surveillance to early detect aneurysm enlargement that could cause rupture. REFERENCES 1. Tsutsumi K, Inoue Y, Hashizume K, et al. Chronic occlusion of an abdominal aortic aneurysm. Ann Vasc Dis 2010;3: 240e3. 2. Sincos IR, da Silva ES, Ragazzo L, et al. Chronic thrombosed abdominal aortic aneurysms: a report on three consecutive cases and literature review. Clinics (Sao Paulo) 2009;64: 1227e30. 3. Dalal S, Donlon M, Beard JD. Thrombosed abdominal aortic aneurysms. Do they need surveillance to prevent late rupture? Eur J Vasc Endovasc Surg 2001;22:570e2.
Annals of Vascular Surgery
4. Wu CY, Rectenwald JE. Incidental discovery of a chronically thrombosed abdominal aortic aneurysm: case report and literature review. Ann Vasc Surg 2015;29:1018.e1e4. 5. Labruto F, Blomqvist L, Swedenborg J. Imaging the intraluminal thrombus of abdominal aortic aneurysms: techniques, findings, and clinical implications review: imaging of intraluminal thrombus in AAA. J Vasc Interv Radiol 2011;22: 1069e75. 6. Schurink GW, Van Baalen JM, Visser MJ, et al. Thrombus within an aortic aneurysm does not reduce pressure on the aneurysmal wall. J Vasc Surg 2000;31:501e6. 7. Takagi H, Yoshikawa S, Mizuno Y, et al. Intrathrombotic pressure of a thrombosed abdominal aortic aneurysm. Ann Vasc Surg 2005;19:108e12. 8. Filis KA, Lagoudianakis EE, Markogiannakis H, et al. Complete abdominal aortic aneurysm thrombosis and obstruction of both common iliac arteries with intrathrombotic pressures demonstrating a continuing risk of rupture: a case report and review of the literature. J Med Case Rep 2009;3:9292. 9. Schwartz RA, Nichols WK, Silver D. Is thrombosis of the infrarenal abdominal aneurysm an acceptable alternative? J Vasc Surg 1986;3:448e55. 10. Parry DJ, Al-Barjas HS, Chappell L, et al. Markers of inflammation in men with small abdominal aortic aneurysm. J Vasc Surg 2010;52:145e51. 11. Bluestein D, Dumont K, De Beule M, et al. Intraluminal thrombus and risk of rupture in patient specific abdominal aortic aneurysm - FSI modelling. Comput Methods Biomech Biomed Engin 2009;12:73e81. 12. Ricotta JJ, Kirshner RL. Case report: late rupture of thrombosed abdominal aortic aneurysm. Surgery 1984;95: 753e5. 13. Kumar V. Endovascular treatment of an acutely thrombosed AAA. J Endovasc Ther 2005;12:70e3. 14. Patel H, Krishnamoorthy M, Dorazio RA, et al. Thrombosis of abdominal aortic aneurysms. Am Surg 1994;60:801e3. 15. Tanaka S, Tanaka K, Morioka K, et al. Thrombosed abdominal aortic aneurysm associated with an extensively ‘‘shaggy’’ aorta repaired anatomically via a thoracoabdominal approach with supraceliac aortic clamping: report of a case. Surg Today 2010;40:866e70. 16. Leather RP, Shah D, Goldman M, et al. Nonresective treatment of abdominal aortic aneurysm. Use of acute thrombosis and axillofemoral bypass. Arch Surg 1979;114: 1402e8.
Chronic totally thrombosed abdominal aortic aneurysms (AAAs) comprise a rare medical situation, with only a few cases reported in literature. Optimal management has been controversial, although an early risk for rupture is present. Therefore, we present a rare case of late rupture in a patient with a totally thrombosed AAA, and we discuss proper treatment.
Spontaneous thrombosis of an abdominal aortic aneurysm (AAA) is a rare condition, with totally thrombosed AAAs being associated with a continuous risk for rupture according to some authors.1 Although all the reports in literature refer to an aneurysm rupture within the first year after presentation, no case of late rupture has been reported to date.2 Therefore, we are presenting a rare case of late rupture in a patient with total AAA thrombosis treated originally with axillobifemoral bypass.
CASE REPORT A 69-year-old male patient was referred to our emergency department with a diagnosis of a ruptured AAA. His medical history included: arterial hypertension (not adequately controlled with medical treatment), coronary artery disease treated with coronary artery bypass grafting almost 6 years ago (1 year before the diagnosis of the AAA), chronic heart failure (ejection fracture ¼ 30%), cerebrovascular disease, right-sided internal carotid artery occlusion (under dual antiplatelet treatment), left-sided internal carotid endarterectomy, and tobacco and alcohol
Department of Vascular Surgery, KAT General Hospital, Athens, Greece. Correspondence to: George Galyfos, Department of Vascular Surgery, KAT General Hospital, 2 Nikis Street, Kifisia, 14561, Athens, Greece; E-mail: [email protected] Ann Vasc Surg 2018; 46: 368.e5–368.e8 http://dx.doi.org/10.1016/j.avsg.2017.07.043 Ó 2017 Elsevier Inc. All rights reserved. Manuscript received: June 29, 2017; manuscript accepted: July 25, 2017; published online: 6 September 2017
abuse. Additionally, he had a history of a spontaneously thrombosed AAA (including the common iliac arteries) that presented 5 years ago (original diameter: 5.8 cm; Fig. 1) with symptoms of intermittent claudication. Owing to the severity of the general condition, he had undergone an axillobifemoral bypass in another institution which resolved the symptoms of the peripheral ischemia. However, the patient was lost on follow-up. On admission, the patient was suffering from severe abdominal and back pain worsening for the last 4 days; he was hemodynamically stable, and a pulsatile abdominal mass was palpated during physical examination. The axillobifemoral bypass was patent with palpable peripheral pulses. Regarding diagnostic imaging, the patient underwent a computed tomography angiography (CTA) that revealed a ruptured infrarenal AAA (maximum diameter ¼ 7 cm) and confirmed the patency of the axillobifemoral prosthetic graft (Fig. 2). Considering treatment, the patient underwent an emergency laparotomy. After dissection of the retroperitoneal hematoma, we proceeded to division of the left renal vein, exposure and clamping of the infrarenal aortic neck, excision of the aortic sac, and evacuation of the intraluminal thrombus, and finally, we primarily ligated the aortic neck and bifurcation (double layer of sutures). The distal aorta and aortic bifurcation were occluded, and therefore, no in situ reconstruction was considered an option. During declamping, a hemorrhage of the right renal artery orifice presented due to aortic clamp injury and excessive calcification of the aorta. After suprarenal aortic clamping, the deficit of the arterial wall was sutured successfully and aortic clamping was removed, with blood supply to the right renal artery being preserved. Finally, a flap of greater omentum was sutured on the aortic stump for further protection.
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Annals of Vascular Surgery
Fig. 1. Computed tomography angiography (CTA) showing a totally thrombosed abdominal aortic aneurysm (AAA, arrow) presented 5 years ago.
Fig. 2. Computed tomography angiography (CTA) showing a rupture of the aneurysmal sac (thick arrow) and the patent axillobifemoral bypass (thin arrow).
Postoperatively, the patient was transferred to intensive care unit for further treatment. Postoperative medical treatment included low-molecular-weight heparin prophylaxis, antiplatelet therapy, and intravenous
antibiotics (standard prophylaxis for 3 days). Owing to a persistent diarrheal syndrome (even after discontinuation of the antibiotics), the patient was discharged 25 days after the procedure. His course remains uneventful
Volume 46, January 2018
Case reports 368.e7
Fig. 3. Postoperative computed tomography angiography (CTA) showing the intact aortic stump just below the level of the renal arteries (thick arrow) and the patent axillobifemoral bypass (thin arrow).
almost 2 months after discharge. Postoperative imaging with CTA shows the intact aortic stump just below the level of the renal arteries and patency of the axillobifemoral bypass (Fig. 3).
DISCUSSION Chronic total AAA occlusion is a relatively rare condition, with reported incidence reaching 0.6e2.8%.1,2 Factors that are associated with AAA thrombosis and occlusion include thromboembolic disease, hypercoagulation states, neoplasm, trauma, fever, iliac artery occlusive disease, and states of low flow such as chronic heart failure.3,4 Clinical presentation in chronic cases most commonly includes intermittent claudication. However, acute thrombosis usually presents with acute lower extremities ischemia and/or renal-visceral ischemia or paraplegia, and it is associated with a high mortality risk (almost 50%).2e5 Regarding pathophysiology, experimental studies have concluded that the lack of blood flow within the aneurysm sac does not reduce the pulse pressure of the thrombus to the aneurysmatic wall, and therefore, the risk of rupture remains.6 Takagi et al.7 have compared systematic and intrathrombotic pressure at 3 points within a thrombosed AAA, and they found only a 1e5% pressure decrease of mean intrathrombotic pressure. Moreover, Filis et al.8 have also compared systematic and intrathrombotic pressures within an occluded AAA, and they have found no significant differences. Additionally, rupture could occur in case a thrombosed aortic sac continued to be pressurized
by collateral branches.6,9 However, in our case, the rupture presented later, and this could be probably attributed to partial depressurization of the sac by the axillobifemoral bypass. Finally, all the reports in literature refer to rupture within 1 year after presentation, with a rupture risk reaching up to 15% in 6 months.2,9 Although some authors have advocated that thrombus could protect from rupture by reducing the wall stress, others have suggested that thrombus could cause sac expansion.1 Thrombosis is considered to involve the interaction between the process of inflammation and coagulation that are deteriorated in patients with AAA.10 Furthermore, intraluminal thrombus has been shown to alter the normal pattern of oxygen supply within the aortic wall leading to local hypoxia, wall weakening, and increasing the risk for rupture.8,11 Compared to the thrombusfree wall, the wall covered by thrombus is characterized by apoptotic smooth muscle cells, fragmented elastin fibers, and inflammatory cells.1,8 Therefore, the thrombus itself could affect the structural integrity and stability of the wall, predisposing to rupture. Considering treatment, many authors have selected the conservative management although this has led to rupture in several cases.12 Endovascular repair of a totally thrombosed AAA would be prohibiting as well, and therefore, there are only limited reports in literature.13 Many other strategies have been described in the past such as thrombectomy, aneursymectomy, and aortoiliac/aortofemoral bypass grafting with promising results, although only small case series have been reported in literature as well.14 However, in high-risk
368.e8 Case reports
patients, such as our patient, axillobifemoral bypass would be an acceptable alternative without treating the aneurysm or conducting a laparotomy.15 In the past, some authors have suggested causing thrombosis to a partially thrombosed AAA with simultaneous axillobifemoral bypass, without reporting any late complication associated with the AAA itself.16 However, follow-up reported after such procedures is of limited duration, and no long-term results have been published. Additionally, preoperative evaluation of the patient regarding the original procedure and the decision not to treat the thrombosed AAA definitely is an issue for consideration. Finally, given the chronic occlusion of the aortic bifurcation and the high surgical risk of the patient, no in situ reconstruction was attempted. Furthermore, the ligated aortic stump was covered with omentum for reinforcement.
CONCLUSIONS Chronically thrombosed AAAs are associated with a significant risk for rupture, even in the long term. Therefore, even if such patients have undergone bypass surgery or if they are under anticoagulant treatment, they should be under close surveillance to early detect aneurysm enlargement that could cause rupture. REFERENCES 1. Tsutsumi K, Inoue Y, Hashizume K, et al. Chronic occlusion of an abdominal aortic aneurysm. Ann Vasc Dis 2010;3: 240e3. 2. Sincos IR, da Silva ES, Ragazzo L, et al. Chronic thrombosed abdominal aortic aneurysms: a report on three consecutive cases and literature review. Clinics (Sao Paulo) 2009;64: 1227e30. 3. Dalal S, Donlon M, Beard JD. Thrombosed abdominal aortic aneurysms. Do they need surveillance to prevent late rupture? Eur J Vasc Endovasc Surg 2001;22:570e2.
Annals of Vascular Surgery
4. Wu CY, Rectenwald JE. Incidental discovery of a chronically thrombosed abdominal aortic aneurysm: case report and literature review. Ann Vasc Surg 2015;29:1018.e1e4. 5. Labruto F, Blomqvist L, Swedenborg J. Imaging the intraluminal thrombus of abdominal aortic aneurysms: techniques, findings, and clinical implications review: imaging of intraluminal thrombus in AAA. J Vasc Interv Radiol 2011;22: 1069e75. 6. Schurink GW, Van Baalen JM, Visser MJ, et al. Thrombus within an aortic aneurysm does not reduce pressure on the aneurysmal wall. J Vasc Surg 2000;31:501e6. 7. Takagi H, Yoshikawa S, Mizuno Y, et al. Intrathrombotic pressure of a thrombosed abdominal aortic aneurysm. Ann Vasc Surg 2005;19:108e12. 8. Filis KA, Lagoudianakis EE, Markogiannakis H, et al. Complete abdominal aortic aneurysm thrombosis and obstruction of both common iliac arteries with intrathrombotic pressures demonstrating a continuing risk of rupture: a case report and review of the literature. J Med Case Rep 2009;3:9292. 9. Schwartz RA, Nichols WK, Silver D. Is thrombosis of the infrarenal abdominal aneurysm an acceptable alternative? J Vasc Surg 1986;3:448e55. 10. Parry DJ, Al-Barjas HS, Chappell L, et al. Markers of inflammation in men with small abdominal aortic aneurysm. J Vasc Surg 2010;52:145e51. 11. Bluestein D, Dumont K, De Beule M, et al. Intraluminal thrombus and risk of rupture in patient specific abdominal aortic aneurysm - FSI modelling. Comput Methods Biomech Biomed Engin 2009;12:73e81. 12. Ricotta JJ, Kirshner RL. Case report: late rupture of thrombosed abdominal aortic aneurysm. Surgery 1984;95: 753e5. 13. Kumar V. Endovascular treatment of an acutely thrombosed AAA. J Endovasc Ther 2005;12:70e3. 14. Patel H, Krishnamoorthy M, Dorazio RA, et al. Thrombosis of abdominal aortic aneurysms. Am Surg 1994;60:801e3. 15. Tanaka S, Tanaka K, Morioka K, et al. Thrombosed abdominal aortic aneurysm associated with an extensively ‘‘shaggy’’ aorta repaired anatomically via a thoracoabdominal approach with supraceliac aortic clamping: report of a case. Surg Today 2010;40:866e70. 16. Leather RP, Shah D, Goldman M, et al. Nonresective treatment of abdominal aortic aneurysm. Use of acute thrombosis and axillofemoral bypass. Arch Surg 1979;114: 1402e8.