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Failed Minimally Invasive Staged Treatment of a Giant Symptomatic Aortic Perigraft Hygroma after Open Aortic Repair
Case Report Failed Minimally Invasive Staged Treatment of a Giant Symptomatic Aortic Perigraft Hygroma after Open Aortic Repair Andres Reyes Valdivia,1 Africa Duque Santos,1 Francisco Alvarez Marcos,2 ~iga,1 Alvaro Osorio Ruiz,1 Julia Oca~ na Guaita,1 and Claudio Gandarias Z un Madrid and A Coru~ na, Spain
Background: Perigraft hygromas or seromas are an unusual finding and/or complication after open aortic repair. Methods and Results: We present a case of an 82-year-old man with a previous urgent aortic bifurcated graft for abdominal aortic aneurysm rupture. He received several treatments due to abdominal compartment syndrome, requiring a Bogota Bag and colostomy derivation. He was finally discharged home and lost on follow-up. Eight years after this procedure, he presented to the urgency department with an abdominal mass and pain. Urgent computed tomography (CT) scan revealed a giant bilobed aortic sac, corresponding with a huge hygroma. A 3-stage minimally invasive procedure was scheduled due to hostile abdomen. Six months after successful treatment, patient came with fever and abdominal pain. He was diagnosed with graft infection and aortoenteric fistula and was treated with explantation and silver in situ repair. Conclusions: Aortic hygroma or seromas after open repair should be treated by open means whenever possible. Endovascular techniques could be a valid option in selected patients; however, further evidence is needed.
Perigraft hygromas (PGH) are an unusual complication of open aortic repair. This complication is mostly encountered with aortic graft procedures; however, PGH have been reported even with vein bypass (1e 2%).1,2 This disease is usually asymptomatic,
This paper was presented at the 30th Annual Meeting of the European Society for Vascular Surgery, Copenhagen, Denmark, September 28e30, 2016, with title ‘‘Failed fully minimally invasive staged treatment of a giant aortic sac aneurysm in extremely hostile abdomen after open repair of a ruptured abdominal aortic aneurysm.’’ 1 Department of Vascular and Endovascular Surgery, Ramon y Cajal’s University Hospital, Madrid, Spain. 2 Department of Vascular and Endovascular Surgery, Juan Canalejo’s University Hospital, A Coru~ na, Spain.
Correspondence to: Andres Reyes Valdivia, MD, FEBVS, Department of Vascular and Endovascular Surgery, Ramon y Cajal’s University Hospital, Madrid, Spain; E-mail: [email protected] Ann Vasc Surg 2017; -: 1–5 http://dx.doi.org/10.1016/j.avsg.2016.12.021 Ó 2017 Elsevier Inc. All rights reserved. Manuscript received: October 11, 2016; manuscript accepted: December 18, 2016; published online: - - -
although mass sensation, pressure symptoms, abdominal tenderness, limb ischemia, or even rupture have been described. A wide variety of pathogenic mechanisms have been proposed as the cause of PGH, such as low-grade infection3 or a fibrinolytic milieu retarding sealing of graft pores.4 Several predisposing factors are also detailed, such as low-grade allergy to material,5 hipoalbuminemia, anticoagulation state, or high blood pressure. Treatment is always an issue, as patients with previous aortic repair are considered as high-risk patients. Graft explantation with thrombus and sac material removal, and in situ graft repair are proposed as definitive treatments. However, in very ill, high-risk patients, some reports have been described with successful endovascular aortic repair (EVAR) exclusion and PGH involution.
CASE REPORT An 82-year-old man previously treated with a bifurcated PTFE graft for AAA rupture. During hospitalization, he 1
2 Case Report
Annals of Vascular Surgery
Fig. 1. Panels (A) and (B) show primary diagnosis CT scan with severe proximal calcification. Panel (C) shows angiography with no endoleaks.
Fig. 2. Summary of the staged scheduled procedure.
required several interventions due to abdominal compartment syndrome. He was treated with a Bogota bag and needed a left colectomy with colostomy derivation. Some weeks later, bowel reconstruction was performed. After 1 month and a half, he was discharged home and lost on follow-up. Eight years later, he presented in the urgent department with an abdominal mass and pain. Urgent computed tomography (CT) scan revealed a giant 10 cm bilobed aortic sac. Previous graft proximal anastomosis was severely calcified, as were the iliac accesses. Angiography revealed no leaks. Blood culture tests and white blood cell count (WBCC) were negative for infection. Patient revealed that the abdominal mass had been enlarging over the last 3 years. With this information, PGH was diagnosed. (Fig. 1). The patient was considered as symptomatic and a moderate-high risk of rupture was given. No further examinations were considered and semiurgent treatment was decided. Due to the extremely hostile abdomen, a 3-stage procedure was scheduled. (Fig. 2). Stage 1 consisted of graft relining with bilateral long selfexpandable covered stents and 2 balloonexpandable covered stents to secure the proximal anastomosis of the previous PTFE graft. Second stage consisted of accessory lobe drainage under CT scan guidance. This procedure was successfully performed. Figure 3 summarizes stage 1 and 2. Final stage was performed 2 weeks later, and main lobe drained under CT guidance and was filled with contrasted glue. Main lobe remained stable on
control CT scan. (Fig. 4). However, 9 months after initial procedure, the patient returned with fever and abdominal pain, corresponding with elevated WBCC and inflammatory markers. He mentioned 1 month of slight fever. Urgent CT scan revealed gas in the sac and a communication with the bowel; therefore, graft infection with aortoenteric fistula was diagnosed. He was urgently treated with graft explantation, silver in situ bypass, and bowel repair. (Fig. 5). Patient recovered, was put on culture specific antibiotics due to Staphylococcus epidermidis positive graft and blood cultures, and was finally discharged home after 1 month of hospitalization.
DISCUSSION Risberg et al. coined the term perigraft hygroma for sac expansion in the absence of detectable endoleak.6 Endoleaks are the main reason of aortic sac expansion after EVAR.7 Sac expansion can also occur in the absence of an endoleak through a process of pressure transmission or fluid ultrafiltration across the graft fabric. Factors that adversely affect this process can lead to continued graft porosity and slow leakage of plasma fluid into the perigraft area. The largest series to date reporting on PGH is the one by Kadakol et al.,8
Volume
-, -
2017
Case Report 3
Fig. 3. CT scan showing proximal balloon-expandable stents in the proximal anastomosis and sac puncture of the accessory lobe.
Fig. 4. Image showing drainage of the main lobe.
describing an incidence of 18% (20 of 111 patients). They reported that all the patients with diagnosis of PGH were treated with PTFE grafts, and on multivariate analysis, they identified several factors
associated with PGH development after open AAA repair. The factors are anticoagulation, left flank extraperitoneal exposure, reconstruction with a bifurcated graft, diabetes, and smoking. Reports on
4 Case Report
Annals of Vascular Surgery
Fig. 5. CT scan showing an intrasac gas with growth. Intraoperative sac image.
PGH after dacron graft are scarce.9 The usual presentation of PGH is asymptomatic, whereas some reports have described on symptomatic patients.10,11 Thoo et al.12 reported 5 cases after open AAA repair with PTFE, 2 of which ruptured. Three additional cases of ruptured expanding hygromas were also reported. Endovascular techniques have been described for hygromas after EVAR with successful outcomes.9 A report of 2 cases by Salameh et al.13 with EVAR for PGH after open repair is so far one of the small evidence for this kind of treatment. Open redo repair has been widely described as main option of treatment.14,15 One report16 has described aortic aneurysm wall endarterectomy previous to sac closure as protective for PGH appearance, improving graft incorporation. In our case, an intention for endovascular exclusion was tempted due to the extreme hostile abdomen. Although relining, depressurization with aspiration and sac filling was successfully performed, the infection made the procedure become a failure. This was probably due to the puncturing procedures in an ill patient. Probably, in these patients with previous abdominal surgeries, a translumbar approach could be beneficial. Evacuation and filling of the sac can be discussed, as previous reports just focus on graft relining. Our intention was to relieve the abdominal pain caused by the severely enlarged aneurysmal sac. Decision on foam and glue sac filling was based on cystic pathologies filling experience, given the inflammatory response of the material. Preventive cephalosporin protocol was used. Standard EVAR in this case was not possible due to short distance between the renal arteries and the origin of the bifurcation and the severely calcified anastomosis. This is
the reason we used additional balloon-expandable stents in the proximal anastomosis. We believe that endovascular approach to exclude a PGH after open repair should remain as a valid procedure in high-risk patients, and maybe, relining alone could be enough. However, open redo repair should be done whenever possible and should also remain as the gold standard of treatment in this unusual complication. REFERENCES 1. Blumenberg RM, Gelfand ML, Dale WA. Perigraft seromas complicating arterial grafts. Surgery 1985;97:194e204. 2. Ahn SS, Machleder HI, Gupta R, et al. Perigraft seroma clinical, histologic, and serologic correlates. Am J Surg 1987;154:173e8. 3. LeBlanc J, Albus R, Williams WG, et al. Serous fluid leakage: a complication following the modified Blalock-Taussig shunt. J Thorac Cardiovasc Surg 1984;88:259e62. 4. Risberg B, Delle M, Eriksson E, et al. Aneurysm sac hygroma: a cause of endotension. J Endovasc Ther 2001;8: 447e53. 5. Williams GM. The management of massive ultrafiltration distending the aneurysm sac after abdominal aortic aneurysm repair with a polytetrafluoroethylene aortobiliac graft. J Vasc Surg 1998;28:551e5. 6. Risberg B, Delle M, Lonn L, et al. Management of aneurysm sac hygroma. J Endovasc Ther 2004;11:191e5. 7. Goodney PP, Fillinger MF. The effect of endograft relining on sac expansion after endovascular aneurysm repair with the original-permeability Gore Excluder abdominal aortic aneurysm endoprosthesis. J Vasc Surg 2007;45:686e93. 8. Kadakol AK, Nypaver TJ, Lin JC, et al. Frequency, risk factors, and management of perigraft seroma after open abdominal aortic aneurysm repair. J Vasc Surg 2011;54:637e43. 9. Ryu RK, Palestrant S, Ryu J, et al. Sac hygroma after endovascular abdominal aortic aneurysm repair: successful treatment with endograft relining. Cardiovasc Intervent Radiol 2007;30:488e90.
Volume
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10. Lucas LA, Rodriguez JA, Olsen DM, et al. Symptomatic seroma after open abdominal aortic aneurysm repair. Ann Vasc Surg 2009;23:144e6. 11. Mennander A, Pimenoff G, Heikkinen M, et al. Nonoperative approach to endotension. J Vasc Surg 2005;42:194e9. 12. Thoo CH, Bourke BM, May J. Symptomatic sac enlargement and rupture due to seroma after open abdominal aortic aneurysm repair with polytetrafluoroethylene graft: implications for endovascular repair and endotension. J Vasc Surg 2004;40:1089e94. 13. Salameh MK, Hoballah JJ. Successful endovascular treatment of aneurysm sac hygroma after open abdominal aortic
Case Report 5
aneurysm replacement: a report of two cases. J Vasc Surg 2008;48:457e60. 14. Cuff RF, Thomas JH. Recurrent symptomatic aortic sac seroma after open abdominal aortic aneurysm repair. J Vasc Surg 2005;41:1058e60. 15. Arya N, O’Kane HF, Hannon RJ, et al. Endoleak and endotension following open abdominal aortic aneurysm repair: a report of two cases. Ann Vasc Surg 2005;19:431e3. 16. Wolff T, Koller MT, Eugster T, et al. Endarterectomy of the aneurysm sac in open abdominal aortic aneurysm repair reduces perigraft seroma and improves graft incorporation. World J Surg 2011;35:905e10.
Background: Perigraft hygromas or seromas are an unusual finding and/or complication after open aortic repair. Methods and Results: We present a case of an 82-year-old man with a previous urgent aortic bifurcated graft for abdominal aortic aneurysm rupture. He received several treatments due to abdominal compartment syndrome, requiring a Bogota Bag and colostomy derivation. He was finally discharged home and lost on follow-up. Eight years after this procedure, he presented to the urgency department with an abdominal mass and pain. Urgent computed tomography (CT) scan revealed a giant bilobed aortic sac, corresponding with a huge hygroma. A 3-stage minimally invasive procedure was scheduled due to hostile abdomen. Six months after successful treatment, patient came with fever and abdominal pain. He was diagnosed with graft infection and aortoenteric fistula and was treated with explantation and silver in situ repair. Conclusions: Aortic hygroma or seromas after open repair should be treated by open means whenever possible. Endovascular techniques could be a valid option in selected patients; however, further evidence is needed.
Perigraft hygromas (PGH) are an unusual complication of open aortic repair. This complication is mostly encountered with aortic graft procedures; however, PGH have been reported even with vein bypass (1e 2%).1,2 This disease is usually asymptomatic,
This paper was presented at the 30th Annual Meeting of the European Society for Vascular Surgery, Copenhagen, Denmark, September 28e30, 2016, with title ‘‘Failed fully minimally invasive staged treatment of a giant aortic sac aneurysm in extremely hostile abdomen after open repair of a ruptured abdominal aortic aneurysm.’’ 1 Department of Vascular and Endovascular Surgery, Ramon y Cajal’s University Hospital, Madrid, Spain. 2 Department of Vascular and Endovascular Surgery, Juan Canalejo’s University Hospital, A Coru~ na, Spain.
Correspondence to: Andres Reyes Valdivia, MD, FEBVS, Department of Vascular and Endovascular Surgery, Ramon y Cajal’s University Hospital, Madrid, Spain; E-mail: [email protected] Ann Vasc Surg 2017; -: 1–5 http://dx.doi.org/10.1016/j.avsg.2016.12.021 Ó 2017 Elsevier Inc. All rights reserved. Manuscript received: October 11, 2016; manuscript accepted: December 18, 2016; published online: - - -
although mass sensation, pressure symptoms, abdominal tenderness, limb ischemia, or even rupture have been described. A wide variety of pathogenic mechanisms have been proposed as the cause of PGH, such as low-grade infection3 or a fibrinolytic milieu retarding sealing of graft pores.4 Several predisposing factors are also detailed, such as low-grade allergy to material,5 hipoalbuminemia, anticoagulation state, or high blood pressure. Treatment is always an issue, as patients with previous aortic repair are considered as high-risk patients. Graft explantation with thrombus and sac material removal, and in situ graft repair are proposed as definitive treatments. However, in very ill, high-risk patients, some reports have been described with successful endovascular aortic repair (EVAR) exclusion and PGH involution.
CASE REPORT An 82-year-old man previously treated with a bifurcated PTFE graft for AAA rupture. During hospitalization, he 1
2 Case Report
Annals of Vascular Surgery
Fig. 1. Panels (A) and (B) show primary diagnosis CT scan with severe proximal calcification. Panel (C) shows angiography with no endoleaks.
Fig. 2. Summary of the staged scheduled procedure.
required several interventions due to abdominal compartment syndrome. He was treated with a Bogota bag and needed a left colectomy with colostomy derivation. Some weeks later, bowel reconstruction was performed. After 1 month and a half, he was discharged home and lost on follow-up. Eight years later, he presented in the urgent department with an abdominal mass and pain. Urgent computed tomography (CT) scan revealed a giant 10 cm bilobed aortic sac. Previous graft proximal anastomosis was severely calcified, as were the iliac accesses. Angiography revealed no leaks. Blood culture tests and white blood cell count (WBCC) were negative for infection. Patient revealed that the abdominal mass had been enlarging over the last 3 years. With this information, PGH was diagnosed. (Fig. 1). The patient was considered as symptomatic and a moderate-high risk of rupture was given. No further examinations were considered and semiurgent treatment was decided. Due to the extremely hostile abdomen, a 3-stage procedure was scheduled. (Fig. 2). Stage 1 consisted of graft relining with bilateral long selfexpandable covered stents and 2 balloonexpandable covered stents to secure the proximal anastomosis of the previous PTFE graft. Second stage consisted of accessory lobe drainage under CT scan guidance. This procedure was successfully performed. Figure 3 summarizes stage 1 and 2. Final stage was performed 2 weeks later, and main lobe drained under CT guidance and was filled with contrasted glue. Main lobe remained stable on
control CT scan. (Fig. 4). However, 9 months after initial procedure, the patient returned with fever and abdominal pain, corresponding with elevated WBCC and inflammatory markers. He mentioned 1 month of slight fever. Urgent CT scan revealed gas in the sac and a communication with the bowel; therefore, graft infection with aortoenteric fistula was diagnosed. He was urgently treated with graft explantation, silver in situ bypass, and bowel repair. (Fig. 5). Patient recovered, was put on culture specific antibiotics due to Staphylococcus epidermidis positive graft and blood cultures, and was finally discharged home after 1 month of hospitalization.
DISCUSSION Risberg et al. coined the term perigraft hygroma for sac expansion in the absence of detectable endoleak.6 Endoleaks are the main reason of aortic sac expansion after EVAR.7 Sac expansion can also occur in the absence of an endoleak through a process of pressure transmission or fluid ultrafiltration across the graft fabric. Factors that adversely affect this process can lead to continued graft porosity and slow leakage of plasma fluid into the perigraft area. The largest series to date reporting on PGH is the one by Kadakol et al.,8
Volume
-, -
2017
Case Report 3
Fig. 3. CT scan showing proximal balloon-expandable stents in the proximal anastomosis and sac puncture of the accessory lobe.
Fig. 4. Image showing drainage of the main lobe.
describing an incidence of 18% (20 of 111 patients). They reported that all the patients with diagnosis of PGH were treated with PTFE grafts, and on multivariate analysis, they identified several factors
associated with PGH development after open AAA repair. The factors are anticoagulation, left flank extraperitoneal exposure, reconstruction with a bifurcated graft, diabetes, and smoking. Reports on
4 Case Report
Annals of Vascular Surgery
Fig. 5. CT scan showing an intrasac gas with growth. Intraoperative sac image.
PGH after dacron graft are scarce.9 The usual presentation of PGH is asymptomatic, whereas some reports have described on symptomatic patients.10,11 Thoo et al.12 reported 5 cases after open AAA repair with PTFE, 2 of which ruptured. Three additional cases of ruptured expanding hygromas were also reported. Endovascular techniques have been described for hygromas after EVAR with successful outcomes.9 A report of 2 cases by Salameh et al.13 with EVAR for PGH after open repair is so far one of the small evidence for this kind of treatment. Open redo repair has been widely described as main option of treatment.14,15 One report16 has described aortic aneurysm wall endarterectomy previous to sac closure as protective for PGH appearance, improving graft incorporation. In our case, an intention for endovascular exclusion was tempted due to the extreme hostile abdomen. Although relining, depressurization with aspiration and sac filling was successfully performed, the infection made the procedure become a failure. This was probably due to the puncturing procedures in an ill patient. Probably, in these patients with previous abdominal surgeries, a translumbar approach could be beneficial. Evacuation and filling of the sac can be discussed, as previous reports just focus on graft relining. Our intention was to relieve the abdominal pain caused by the severely enlarged aneurysmal sac. Decision on foam and glue sac filling was based on cystic pathologies filling experience, given the inflammatory response of the material. Preventive cephalosporin protocol was used. Standard EVAR in this case was not possible due to short distance between the renal arteries and the origin of the bifurcation and the severely calcified anastomosis. This is
the reason we used additional balloon-expandable stents in the proximal anastomosis. We believe that endovascular approach to exclude a PGH after open repair should remain as a valid procedure in high-risk patients, and maybe, relining alone could be enough. However, open redo repair should be done whenever possible and should also remain as the gold standard of treatment in this unusual complication. REFERENCES 1. Blumenberg RM, Gelfand ML, Dale WA. Perigraft seromas complicating arterial grafts. Surgery 1985;97:194e204. 2. Ahn SS, Machleder HI, Gupta R, et al. Perigraft seroma clinical, histologic, and serologic correlates. Am J Surg 1987;154:173e8. 3. LeBlanc J, Albus R, Williams WG, et al. Serous fluid leakage: a complication following the modified Blalock-Taussig shunt. J Thorac Cardiovasc Surg 1984;88:259e62. 4. Risberg B, Delle M, Eriksson E, et al. Aneurysm sac hygroma: a cause of endotension. J Endovasc Ther 2001;8: 447e53. 5. Williams GM. The management of massive ultrafiltration distending the aneurysm sac after abdominal aortic aneurysm repair with a polytetrafluoroethylene aortobiliac graft. J Vasc Surg 1998;28:551e5. 6. Risberg B, Delle M, Lonn L, et al. Management of aneurysm sac hygroma. J Endovasc Ther 2004;11:191e5. 7. Goodney PP, Fillinger MF. The effect of endograft relining on sac expansion after endovascular aneurysm repair with the original-permeability Gore Excluder abdominal aortic aneurysm endoprosthesis. J Vasc Surg 2007;45:686e93. 8. Kadakol AK, Nypaver TJ, Lin JC, et al. Frequency, risk factors, and management of perigraft seroma after open abdominal aortic aneurysm repair. J Vasc Surg 2011;54:637e43. 9. Ryu RK, Palestrant S, Ryu J, et al. Sac hygroma after endovascular abdominal aortic aneurysm repair: successful treatment with endograft relining. Cardiovasc Intervent Radiol 2007;30:488e90.
Volume
-, -
2017
10. Lucas LA, Rodriguez JA, Olsen DM, et al. Symptomatic seroma after open abdominal aortic aneurysm repair. Ann Vasc Surg 2009;23:144e6. 11. Mennander A, Pimenoff G, Heikkinen M, et al. Nonoperative approach to endotension. J Vasc Surg 2005;42:194e9. 12. Thoo CH, Bourke BM, May J. Symptomatic sac enlargement and rupture due to seroma after open abdominal aortic aneurysm repair with polytetrafluoroethylene graft: implications for endovascular repair and endotension. J Vasc Surg 2004;40:1089e94. 13. Salameh MK, Hoballah JJ. Successful endovascular treatment of aneurysm sac hygroma after open abdominal aortic
Case Report 5
aneurysm replacement: a report of two cases. J Vasc Surg 2008;48:457e60. 14. Cuff RF, Thomas JH. Recurrent symptomatic aortic sac seroma after open abdominal aortic aneurysm repair. J Vasc Surg 2005;41:1058e60. 15. Arya N, O’Kane HF, Hannon RJ, et al. Endoleak and endotension following open abdominal aortic aneurysm repair: a report of two cases. Ann Vasc Surg 2005;19:431e3. 16. Wolff T, Koller MT, Eugster T, et al. Endarterectomy of the aneurysm sac in open abdominal aortic aneurysm repair reduces perigraft seroma and improves graft incorporation. World J Surg 2011;35:905e10.