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Cholesterol Crystal Embolism and Delayed-onset Paraplegia after Thoracoabdominal Aneurysm Repair
Case Report Cholesterol Crystal Embolism and Delayedonset Paraplegia after Thoracoabdominal Aneurysm Repair Bahaa Nasr,1 Fabrice Schneider,2 Pedro Marques da Fonseca,1 and Pierre Gouny,1 Brest and Poitiers, France
Postoperative paraplegia caused by ischemic injury of the spinal cord is the most disabling complication of thoracoabdominal surgery. We described the case of a 75-year-old patient who underwent a thoracoabdominal aneurysm repair (type IV aneurysm according to Crawford classification). The aorta clamping was done at the T11 level without specific medullary protection. The first postoperative week was uneventful. On the postoperative day 8, renal failure and livedo of the left lower limb occurred together with complete hypotonic paraplegia and severe sepsis. Exploratory laparotomy revealed a gangrenous cholecystitis, and skin biopsies showed cholesterol crystals embolisms in the hypodermis small arteries. The patient died on the postoperative day 28 from a multiorgan failure. In this case, paraplegia was due to cholesterol crystal embolism, which migrated secondarily after aortic clamping.
Delayed neurologic deficit, paraplegia, and paraparesis, are the most devastating complications after repair of a descending thoracic or thoracoabdominal aortic aneurysm (TAA).1 Spinal cord ischemia during TAA repair can result from aortic cross clamping by embolism or ischemia and insufficient restoration of the spinal cord blood flow thereafter. We describe here the case of one patient with delayed neurologic deficit due to cholesterol crystal embolism.
CASE REPORT A 75-year-old man has been hospitalized for open repair of a thoracoabdominal aneurysm. The patient was 1 Department of Thoracic and Cardiovascular Surgery, University Hospital of Brest, Brest, France. 2 Department of Thoracic and Vascular Surgery, University Hospital of Poitiers, Poitiers, France.
Correspondence to: Bahaa Nasr, MD, Service de chirurgie cardiaque thoracique et vasculaire, CHU BresteCavale Blanche, Bd Tanguy Prigent, 29609 BresteCedex, France; E-mail: [email protected] Ann Vasc Surg 2014; -: 1–3 http://dx.doi.org/10.1016/j.avsg.2013.11.020 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: July 6, 2013; manuscript accepted: November 28, 2013; published online: ---.
diabetic and heavy active smoker. A cardiac ultrasonography revealed an ischemic heart disease with an anterior myocardial necrosis. Ejection fraction was 52% on the isotopic ventriculography. Aortography showed a large thoracoabdominal aneurysm extending from T12, above the level of the celiac trunk, to the aortic bifurcation (type IV TAA). The computed tomography (CT) scan showed a thoracoabdominal aneurysm of 70 mm diameter with a chronic posterior rupture. Above the clamping area, the aorta is considered to be healthy on the CT scan done preoperatively. The selective spinal cord angiography showed the emergence of the Adamkiewicz artery at the T10 level (Fig. 1). The operation was performed through a left thoracolombotomy. The TAA extended from the celiac trunk to the aortic bifurcation. The aorta was clamped at the T11 level without any medullary protection because of the fact that the origin of Adamkiewicz artery was seen at T10 level above the proximal aortic clamping zone. At the opening of the aneurysm, the posterior rupture was confirmed. The atheromatous fragments and the intraaneurysmal thrombus were examined on anatomopathology, showing the presence of thrombus containing cholesterol crystals. The procedure was carried out according to Crawford method. The proximal anastomosis was done in an end-to-end fashion with a 26-mm diameter polyester graft with reimplantation of the visceral arteries by means of an aortic patch. The distal anastomosis was performed end-to-end
1
2 Case Report
Annals of Vascular Surgery
Fig. 3. Small arteries crystal cholesterol embolism.
Fig. 1. Selective spinal cord angiography.
graft, no collection, or leakage at the level of the anastomoses (the prosthesis was implanted on the T11eT12 junction). At the clamping zone, the CT scan showed an almost circumferential thrombus (Fig. 2). This area did not exist on the CT scan done preoperatively. A cholecystectomy was done. The gallbladder was gangrenous, but there was no peritonitis, liver, or bowel ischemia. The paraplegia remained unchanged, and the patient went on hemodialysis every 3 days but died on the 28th day from a multiorgan failure. Necropsy was not realized. The presence of cholesterol crystals embolism was proved on skin biopsies (Fig. 3), histology of gangrenous gallbladder, and histology of the aortic thrombus withdrawn intraoperatively. Sepsis had a double etiology (gallbladder and lung). There was no hemodynamic complication requiring treatment with vasopressors.
DISCUSSION
Fig. 2. Postoperative CT scan: the circumferential thrombus.
at the level of the aortic bifurcation. The total aorta clamping time including both aortic anastomoses and visceral arteries reimplantation was 56 min. The patient awoke without any neurologic deficit, and he was extubated at day 2. He was reintubated at day 5 because of hypoxemia. He remained hemodynamically stable with an arterial pressure of 150/90 mm Hg. On the eighth postoperative day, the patient presented with acute renal failure, creatinine clearance ¼ 7 mL/min, livedo of both lower limbs and left blue toe, complete hypotonic paraplegia, and a sepsis with 22.900 white blood cells. The blood culture was negative. However, bronchic aspiration fluid cultures were positive for Acinobacter baumani, Staphylococcus coagulase positive, and nonb^eta hemolytic Streptococcus. The abdominal ultrasonography revealed the presence of acalculous cholecystitis. An angio CT showed a patent
Spinal cord ischemia remains a major complication after TAA open surgery.1,2 The new fenestrated endovascular treatment is technically difficult in type IV TAA,3 and surgery remains the gold standard in 2012. The Crawford method requires a double thoracic and abdominal incision and massive transfusion. From a clinical point of view, the anterior spinal artery syndrome consists of a paraplegia associated with altered thermoalgesic sensitivity. The tactile sensitivity may be less altered, and the deep sensitivity remains intact. This complication may occur while the patient is still in the recovery room or in the days after the surgery. Etz et al.4 have reported a series of 90 patients operated of a thoracoabdominal aneurysm. The early mortality rate was 11.1%, and the neurologic complications rate was 15%. The aneurysm repair with visceral and intercostal arteries reimplantation according to Crawford method was and is still considered the usual method of aortic restoration.5 In our case, there was no
Vol.
-,
No.
-, -
2014
reimplantation because of the fact that the origin of the Adamkiewicz artery was above the proximal aortic clamping zone. The main factors that induce spinal cord injury are: 1. Prolonged medullary ischemia due to insufficiency of perfusion during the aortic clampage.2,6 2. Suppression of medullary vascularization by resection or exclusion of an aortic zone including the area of emergence of Adamkiewicz artery.1,2 3. The increase in the pressure of cerebrospinal fluid due to hemodynamic disorders.7 In our case, we do not have any of these factors because the clamping time was <60 min, Adamkiewicz artery origin was above the clamping zone, and there were no intraoperative hemodynamic disorders. The following are the diagnostic criteria of cholesterol embolism: aged >55 years in 76% of cases, severely symptomatic atherosclerotic patients; renal insufficiency, and clinical association of blue toes, livedo, and arterial hypertension. In one-third of the cases, we find a trigger that can be the anticoagulant treatment, fibrinolytics, vascular surgery, an arterial puncture during arteriography, an endoluminal dilatation, or an aortic endoprosthesis procedure. In half of the cases, an abdominal aortic aneurysm is present, and the cholesterol embolism is not diagnosed while the patients are alive. In an animal model of induced ischemic spinal paralysis by direct intraaortic injection of cholesterol suspension, the histologic examination of the spinal cord of the paralyzed rats showed prominent cholesterol crystals blocking the lumen of the anterior and/or the posterior spinal arteries,8 skin biopsy showed embolization of atheromatous plates in the arteries of the subcutaneous tissue. Repeating the skin biopsies and other tissues such as kidney, quadriceps, and iliac bone marrow allows an anatomic confirmation of the clinical diagnosis. In some cases of cholesterol embolism, hyperbaric oxygenation was reported to be efficacious.9 During the thoracoabdominal surgery, the hypoperfusion is the main cause of the spinal cord ischemia but cholesterol embolism may be involved. In our case, the cholesterol emboli diagnosis is based on the following signs: 1. Emergence of the Adamkiewicz artery above the site of the aorta clamping.
Case Report 3
2. Absence of paraplegia during the first 7 postoperative days. 3. Sudden onset of paraplegia associated with general cutaneous signs (livedo). 4. Skin biopsies showing the presence of cholesterol crystal embolism. 5. Absence of usual causes of paraplegia. 6. Concomitant occurrence of a renal failure. 7. Cholesterol embolism was found on the gallbladder biopsy. 8. Appearance of the aorta on the postoperative CT scan (thrombus area near clamping area). 9. The aortic thrombus histology showing the presence of cholesterol emboli. We can plead the presence of cholesterol crystals in the aortic wall, revealed by the aortic clamping level and which have migrated secondarily. REFERENCES 1. Coselli JS, Lemaire SA, Conklin LD, Koksoy C, Schmittling ZC. Morbidity and mortality after extent II thoracoabdominal aortic aneurysm. Ann Thorac Surg 2002;73: 1107e15. 2. Woo EY, Mcgarvey M, Jackson BM, Bavaria JE, Fairman RM, Pochettino A. Spinal cord ischemia may be reduced via a novel technique of intercostal artery revascularization during open thoracoabdominal aneurysm repair. J Vasc Surg 2007;46:421e6. 3. D’Elia P, Tyrrell M, Sobocinski J, Azzaoui R, Koussa M, Haulon S. Endovascular thoracoabdominal aortic aneurysm repair: a literature review of early and mid-term results. J Cardiovasc Surg 2009;50:439e45. 4. ETZ CD, Zoli S, Mueller CS, et al. Staged repair significantly reduces paraplegia rate after extensive thoracoabdominal aortic aneurysm repair. J Thorac Cardiovasc Surg 2010;139: 1464e72. 5. Ross SD, Kron IL, Parrino PE, Shockey KS, Kern JA, Tribble CG. Preservation of intercostal arteries during thoracoabdominal aortic aneurysm surgery: a retrospective study. J Thorac Cardiovasc Surg 1999;118:17e25. 6. Bicknell CD, Riga CV, Wolfe JH. Prevention of paraplegia during thoracoabdominal aortic aneurysm repair. Eur J Vasc Endovasc Surg 2009;37:654e60. 7. Sueda T, Okada K, Watari M, Orihashi K, Shikata H, Matsuura Y. Evaluation of motor and sensory-evoked potentials for spinal cord monitoring during thoraco-abdominal aortic aneurysm surgery. J Thorac Cardiovasc Surg 2000;48: 60e5. 8. Saklayen MG, Goldstein DL, Park YS, Mathews T. Animal model of spinal cord infarction induced by cholesterol embolization. Am J Med Sci 1995;309:49e52. 9. Puttaswamy V, Bennet M, Frawley JE. Hyperbaric oxygenation treatment of acute paraplegia after resection of a thoraco-abdominal aortic aneurysm. J Vasc Surg 1999;30: 1158e61.
Postoperative paraplegia caused by ischemic injury of the spinal cord is the most disabling complication of thoracoabdominal surgery. We described the case of a 75-year-old patient who underwent a thoracoabdominal aneurysm repair (type IV aneurysm according to Crawford classification). The aorta clamping was done at the T11 level without specific medullary protection. The first postoperative week was uneventful. On the postoperative day 8, renal failure and livedo of the left lower limb occurred together with complete hypotonic paraplegia and severe sepsis. Exploratory laparotomy revealed a gangrenous cholecystitis, and skin biopsies showed cholesterol crystals embolisms in the hypodermis small arteries. The patient died on the postoperative day 28 from a multiorgan failure. In this case, paraplegia was due to cholesterol crystal embolism, which migrated secondarily after aortic clamping.
Delayed neurologic deficit, paraplegia, and paraparesis, are the most devastating complications after repair of a descending thoracic or thoracoabdominal aortic aneurysm (TAA).1 Spinal cord ischemia during TAA repair can result from aortic cross clamping by embolism or ischemia and insufficient restoration of the spinal cord blood flow thereafter. We describe here the case of one patient with delayed neurologic deficit due to cholesterol crystal embolism.
CASE REPORT A 75-year-old man has been hospitalized for open repair of a thoracoabdominal aneurysm. The patient was 1 Department of Thoracic and Cardiovascular Surgery, University Hospital of Brest, Brest, France. 2 Department of Thoracic and Vascular Surgery, University Hospital of Poitiers, Poitiers, France.
Correspondence to: Bahaa Nasr, MD, Service de chirurgie cardiaque thoracique et vasculaire, CHU BresteCavale Blanche, Bd Tanguy Prigent, 29609 BresteCedex, France; E-mail: [email protected] Ann Vasc Surg 2014; -: 1–3 http://dx.doi.org/10.1016/j.avsg.2013.11.020 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: July 6, 2013; manuscript accepted: November 28, 2013; published online: ---.
diabetic and heavy active smoker. A cardiac ultrasonography revealed an ischemic heart disease with an anterior myocardial necrosis. Ejection fraction was 52% on the isotopic ventriculography. Aortography showed a large thoracoabdominal aneurysm extending from T12, above the level of the celiac trunk, to the aortic bifurcation (type IV TAA). The computed tomography (CT) scan showed a thoracoabdominal aneurysm of 70 mm diameter with a chronic posterior rupture. Above the clamping area, the aorta is considered to be healthy on the CT scan done preoperatively. The selective spinal cord angiography showed the emergence of the Adamkiewicz artery at the T10 level (Fig. 1). The operation was performed through a left thoracolombotomy. The TAA extended from the celiac trunk to the aortic bifurcation. The aorta was clamped at the T11 level without any medullary protection because of the fact that the origin of Adamkiewicz artery was seen at T10 level above the proximal aortic clamping zone. At the opening of the aneurysm, the posterior rupture was confirmed. The atheromatous fragments and the intraaneurysmal thrombus were examined on anatomopathology, showing the presence of thrombus containing cholesterol crystals. The procedure was carried out according to Crawford method. The proximal anastomosis was done in an end-to-end fashion with a 26-mm diameter polyester graft with reimplantation of the visceral arteries by means of an aortic patch. The distal anastomosis was performed end-to-end
1
2 Case Report
Annals of Vascular Surgery
Fig. 3. Small arteries crystal cholesterol embolism.
Fig. 1. Selective spinal cord angiography.
graft, no collection, or leakage at the level of the anastomoses (the prosthesis was implanted on the T11eT12 junction). At the clamping zone, the CT scan showed an almost circumferential thrombus (Fig. 2). This area did not exist on the CT scan done preoperatively. A cholecystectomy was done. The gallbladder was gangrenous, but there was no peritonitis, liver, or bowel ischemia. The paraplegia remained unchanged, and the patient went on hemodialysis every 3 days but died on the 28th day from a multiorgan failure. Necropsy was not realized. The presence of cholesterol crystals embolism was proved on skin biopsies (Fig. 3), histology of gangrenous gallbladder, and histology of the aortic thrombus withdrawn intraoperatively. Sepsis had a double etiology (gallbladder and lung). There was no hemodynamic complication requiring treatment with vasopressors.
DISCUSSION
Fig. 2. Postoperative CT scan: the circumferential thrombus.
at the level of the aortic bifurcation. The total aorta clamping time including both aortic anastomoses and visceral arteries reimplantation was 56 min. The patient awoke without any neurologic deficit, and he was extubated at day 2. He was reintubated at day 5 because of hypoxemia. He remained hemodynamically stable with an arterial pressure of 150/90 mm Hg. On the eighth postoperative day, the patient presented with acute renal failure, creatinine clearance ¼ 7 mL/min, livedo of both lower limbs and left blue toe, complete hypotonic paraplegia, and a sepsis with 22.900 white blood cells. The blood culture was negative. However, bronchic aspiration fluid cultures were positive for Acinobacter baumani, Staphylococcus coagulase positive, and nonb^eta hemolytic Streptococcus. The abdominal ultrasonography revealed the presence of acalculous cholecystitis. An angio CT showed a patent
Spinal cord ischemia remains a major complication after TAA open surgery.1,2 The new fenestrated endovascular treatment is technically difficult in type IV TAA,3 and surgery remains the gold standard in 2012. The Crawford method requires a double thoracic and abdominal incision and massive transfusion. From a clinical point of view, the anterior spinal artery syndrome consists of a paraplegia associated with altered thermoalgesic sensitivity. The tactile sensitivity may be less altered, and the deep sensitivity remains intact. This complication may occur while the patient is still in the recovery room or in the days after the surgery. Etz et al.4 have reported a series of 90 patients operated of a thoracoabdominal aneurysm. The early mortality rate was 11.1%, and the neurologic complications rate was 15%. The aneurysm repair with visceral and intercostal arteries reimplantation according to Crawford method was and is still considered the usual method of aortic restoration.5 In our case, there was no
Vol.
-,
No.
-, -
2014
reimplantation because of the fact that the origin of the Adamkiewicz artery was above the proximal aortic clamping zone. The main factors that induce spinal cord injury are: 1. Prolonged medullary ischemia due to insufficiency of perfusion during the aortic clampage.2,6 2. Suppression of medullary vascularization by resection or exclusion of an aortic zone including the area of emergence of Adamkiewicz artery.1,2 3. The increase in the pressure of cerebrospinal fluid due to hemodynamic disorders.7 In our case, we do not have any of these factors because the clamping time was <60 min, Adamkiewicz artery origin was above the clamping zone, and there were no intraoperative hemodynamic disorders. The following are the diagnostic criteria of cholesterol embolism: aged >55 years in 76% of cases, severely symptomatic atherosclerotic patients; renal insufficiency, and clinical association of blue toes, livedo, and arterial hypertension. In one-third of the cases, we find a trigger that can be the anticoagulant treatment, fibrinolytics, vascular surgery, an arterial puncture during arteriography, an endoluminal dilatation, or an aortic endoprosthesis procedure. In half of the cases, an abdominal aortic aneurysm is present, and the cholesterol embolism is not diagnosed while the patients are alive. In an animal model of induced ischemic spinal paralysis by direct intraaortic injection of cholesterol suspension, the histologic examination of the spinal cord of the paralyzed rats showed prominent cholesterol crystals blocking the lumen of the anterior and/or the posterior spinal arteries,8 skin biopsy showed embolization of atheromatous plates in the arteries of the subcutaneous tissue. Repeating the skin biopsies and other tissues such as kidney, quadriceps, and iliac bone marrow allows an anatomic confirmation of the clinical diagnosis. In some cases of cholesterol embolism, hyperbaric oxygenation was reported to be efficacious.9 During the thoracoabdominal surgery, the hypoperfusion is the main cause of the spinal cord ischemia but cholesterol embolism may be involved. In our case, the cholesterol emboli diagnosis is based on the following signs: 1. Emergence of the Adamkiewicz artery above the site of the aorta clamping.
Case Report 3
2. Absence of paraplegia during the first 7 postoperative days. 3. Sudden onset of paraplegia associated with general cutaneous signs (livedo). 4. Skin biopsies showing the presence of cholesterol crystal embolism. 5. Absence of usual causes of paraplegia. 6. Concomitant occurrence of a renal failure. 7. Cholesterol embolism was found on the gallbladder biopsy. 8. Appearance of the aorta on the postoperative CT scan (thrombus area near clamping area). 9. The aortic thrombus histology showing the presence of cholesterol emboli. We can plead the presence of cholesterol crystals in the aortic wall, revealed by the aortic clamping level and which have migrated secondarily. REFERENCES 1. Coselli JS, Lemaire SA, Conklin LD, Koksoy C, Schmittling ZC. Morbidity and mortality after extent II thoracoabdominal aortic aneurysm. Ann Thorac Surg 2002;73: 1107e15. 2. Woo EY, Mcgarvey M, Jackson BM, Bavaria JE, Fairman RM, Pochettino A. Spinal cord ischemia may be reduced via a novel technique of intercostal artery revascularization during open thoracoabdominal aneurysm repair. J Vasc Surg 2007;46:421e6. 3. D’Elia P, Tyrrell M, Sobocinski J, Azzaoui R, Koussa M, Haulon S. Endovascular thoracoabdominal aortic aneurysm repair: a literature review of early and mid-term results. J Cardiovasc Surg 2009;50:439e45. 4. ETZ CD, Zoli S, Mueller CS, et al. Staged repair significantly reduces paraplegia rate after extensive thoracoabdominal aortic aneurysm repair. J Thorac Cardiovasc Surg 2010;139: 1464e72. 5. Ross SD, Kron IL, Parrino PE, Shockey KS, Kern JA, Tribble CG. Preservation of intercostal arteries during thoracoabdominal aortic aneurysm surgery: a retrospective study. J Thorac Cardiovasc Surg 1999;118:17e25. 6. Bicknell CD, Riga CV, Wolfe JH. Prevention of paraplegia during thoracoabdominal aortic aneurysm repair. Eur J Vasc Endovasc Surg 2009;37:654e60. 7. Sueda T, Okada K, Watari M, Orihashi K, Shikata H, Matsuura Y. Evaluation of motor and sensory-evoked potentials for spinal cord monitoring during thoraco-abdominal aortic aneurysm surgery. J Thorac Cardiovasc Surg 2000;48: 60e5. 8. Saklayen MG, Goldstein DL, Park YS, Mathews T. Animal model of spinal cord infarction induced by cholesterol embolization. Am J Med Sci 1995;309:49e52. 9. Puttaswamy V, Bennet M, Frawley JE. Hyperbaric oxygenation treatment of acute paraplegia after resection of a thoraco-abdominal aortic aneurysm. J Vasc Surg 1999;30: 1158e61.