Case Report Abdominal Aortic Rupture and Spondylodiscitis: Emergent EVAR and Staged Spinal Fixation Laura Capoccia,1 Danilo Menna,1 Nunzio Montelione,1 Roberto Tarantino,2 Daniele Marruzzo,2 Enrico Sbarigia,1 and Francesco Speziale,1 Rome, Italy
Concomitant aortic aneurismal pathology and vertebral erosion are seldom reported in literature. The differential diagnosis between a primary vertebral disease affecting the aortic wall and a primary aortic pathology causing a vertebral disruption is quite difficult. We report on a patient presenting with increasing lumbar pain and neurologic lower limbs deficit due to a vertebral erosion accompanied by aortic rupture treated by emergent endovascular aortic repair procedure and then staged vertebral fixation. Microbiological tests on intraoperative periaortic fluid collection samples showed no clear sign of infection and clinical conditions progressively improved. At 12-month follow-up, the patient is in good clinical condition, with a small residual walking impairment and no clinical, laboratory, or imaging sign of aortic endograft infection.
Concomitant aortic aneurismal pathology and vertebral erosion are seldom reported in literature.1 These 2 conditions can be diagnosed simultaneously because of the presenting symptoms related to the vertebral disruption, such as lower limb neurologic deficit or lumbar pain, or rarely because of the presence of an abdominal pulsating mass accompanied by slight back pain.2 The differential diagnosis between a primary vertebral disease affecting the aortic wall and a primary aortic pathology causing a vertebral disruption is quite difficult. Preferred diagnostic imaging modalities are contrastenhanced computed tomography (CT) scan and magnetic resonance imaging (MRI), that can help
1 ‘‘Paride Stefanini’’ Department, Vascular and Endovascular Surgery Division, Sapienza University of Rome. 2 Roberto Tarantino and Daniele Marruzzo are neurosurgeons referring to Neurosurgery Division, Sapienza University of Rome.
Correspondence to: Danilo Menna, MD, Viale del Policlinico 155, 00161 Rome, Italy. Tel: +393932667879; fax: +39064940532.; E-mails:
[email protected] or
[email protected] Ann Vasc Surg 2014; -: 1–5 http://dx.doi.org/10.1016/j.avsg.2013.08.029 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: July 12, 2013; manuscript accepted: August 14, 2013; published online: ---.
identifying some pathologic features of the 2 involved districts.3 The pathognomonic features could sometimes lead to the diagnosis of Behc¸et’s disease, Pott’s disease, or syphilitic aortitis, that are, nonetheless, rarely reported in literature.4,5 We report on a patient presenting with increasing lumbar pain and neurologic lower limbs deficit due to a vertebral erosion accompanied by aortic rupture treated by emergent endovascular aortic repair (EVAR) procedure and then staged vertebral fixation.
CASE REPORT A 75-yr-old man, heavy smoker, affected by hypertension, with a past medical history of myocardial infarction and L3eL4 discectomy for lumbar hernia performed 25 yr before, was admitted at our emergency department because of an increasing lumbar pain dramatically worsened in the last 2 days. He reported a progressive reduction of walking distance in the past 3 months, with a sudden worsening of neurologic sensory and motor lower limbs deficit in the last 2 days. On hospital admission, he had no fever, and his blood pressure was 100/80 mm Hg. At physical examination, he presented a small pulsating abdominal mass, Lasegue’s sign positivity on the left side, and all peripheral pulses were present. At serum 1
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Fig. 1. Abdominal magnetic resonance imaging and contrast-enhanced computed tomographic scans detecting the aortic rupture, surrounding hemorrhage, periaortic fluid collection, and L5 vertebral erosion.
Fig. 2. Aortic contrast-enhanced computed tomographic scan and intraoperative angiography showing the aortic rupture.
laboratory tests, hemoglobin level was 9.3 g/dL, with normal creatinine, white blood cells, and platelet count levels. He was promptly submitted to enhanced MRI and CT scans that revealed an infrarenal aortic rupture near the bifurcation without any aneurysmal dilatation (mean infrarenal aortic diameter was 23 mm), a wide retroperitoneal hematoma involving the left psoas muscle, and an erosion of L4eL5 vertebral bodies accompanied by a perivertebral and periaortic fluid collection (Fig. 1). As the patient became hemodynamically unstable, an emergency EVAR procedure was planned to treat the aortic rupture (Fig. 2). The intervention was performed under local anesthesia after surgical exposure of both femoral axes. A Gore Excluder C3 26-14-14 endograft (W.L. Gore & Associates, Inc., Flagstaff, AZ) was deployed, and no collateral vessel embolization was performed. Operating time was about 55 min and 50 cc of low-osmolarity iodinated contrast medium (Iomeron 320 mg/mL, Bracco, Milano, Italy) was administered. In the postoperative period, the patient recovered well, neither hemodynamic instability nor fever was
detected, the back pain progressively improved, but he complained about a persistent lower limbs neurologic sensory and motor deficit (more severe on the left side). The patient was administered intravenous antibiotic therapy (piperacillin + tazobactam 4 g + 500 mg 3 times a day, rifampin 600 mg/day, and fluconazole 200 mg/day) together with his scheduled medicaments. Serum tests progressively went back to normal. Postoperative enhanced CT scans showed a low-flow type II endoleak originating from 2 lumbar arteries causing a small persistent supply of the hematoma through the aortic rupture, even with no hemoglobin level decrease in the following days. The patient underwent a neurologic consult for vertebral stabilization and a staged spinal fixation was planned. Through a posterior access, a decompressive laminectomy was performed followed by pedicle screw fixation involving the spine tract between D11 and ileum. Seven days after the posterior fixation, through left lombotomy and extraperitoneal access, the retroperitoneal hematoma was removed and samples from vertebral
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Fig. 3. Intraoperative pictures showing the hematoma drainage (left), the endograft visible through the aortic rupture (center), and the lumbar arteries ligation (right) through left lombotomy and extraperitoneal access.
Fig. 4. Intraoperative expandable titanium vertebral cage implantation (left) and postoperative scan (right) showing anterior and posterior vertebral stabilization. bone tissue, periaortic fluid, and aortic wall fragments were collected and analyzed (Fig. 3). Intraoperatively, all the aortic supplying lumbar arteries were sutured, then a vertebral L3eL4eL5 corpectomy was performed, and an expandable titanium vertebral cage was implanted (Fig. 4). Intraoperatively, no clearly purulent collection was detected. Postoperative course was uneventful. Six days after spine repair, the patient underwent a further enhanced CT scan that revealed optimal exclusion of the aortic rupture and resolution of type II endoleak with no persistent hematoma. Microbiological tests such as WidaleWright test and GeneXpert System MTB/RIF were negative, whereas Quantiferon-TB Gold test was positive. In the postoperative period, the patient underwent brace treatment for 3 months, intravenous antibiotic therapy for 1 month (piperacillin + tazobactam 4 g + 500 mg 3 times a day) followed by oral antibiotic therapy for 4 months (amoxicillin + clavulanic acid 875 mg + 125 mg/day), until normal levels of inflammatory serum markers were detected.
The patient was evaluated at 1, 3, 6, and 9 months from discharge using blood tests and ultrasonography, with none of these examinations showing signs suggestive for local or systemic infection. At 12-months follow-up, the patient is in good clinical condition, with a small residual walking impairment and no clinical, laboratory, or imaging sign of aortic endograft infection (Fig. 5).
DISCUSSION Vertebral erosion is mainly caused by neoplastic or infectious diseases. It can be sometimes associated with aortic wall disorders, such as aneurysms or contained rupture. The chronic-contained rupture of an abdominal aortic aneurysm is relatively rare (2.7% of operated infrarenal aortic aneurysms).6 The mechanism of chronic-contained abdominal aortic
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Fig. 5. Post-EVAR contrast-enhanced computed tomographic scans before (left side) and after (right side) open surgical ligation of lumbar arteries. The type II endoleak
by lumbar arteries was no more detectable after surgical ligation (right side images).
aneurysm rupture is not clear. A dual mechanism has been postulated7; in small aneurysms, a limited tear can be contained by a still relatively strong wall; in large aneurysms, the size of the sac can promote a high perianeurysmal reaction. Some unrecognized biochemical mechanisms surely can play a role in differentiating those aneurysms prone to sudden rupture from those that can develop a contained disruption. The preferential site of disruption in contained ruptures is the posterior wall, because the strength of the vertebrae can tamponade the blood leakage. In a report by Galessiere et al.8, a vertebral erosion was encountered in 25% of contained ruptures. From a report by Booth et al.9 published in 2002, a retroperitoneal hematoma after an aortic rupture was found to lead to vertebral erosion in 30% of the cases.2 Differential diagnosis between an erosion caused by an aneurysm and erosion caused by an infection is not always simple, because symptoms related to an aortic disease can be sometimes subtle and usually include back pain and/or neurologic lower limbs motor or sensory deficit. In the reported case, the
patient complained about a progressive reduction of walking distance in the past 3 months, with a sudden worsening 2 days before hospital admission. Preferred diagnostic modalities in those cases are CT and MRI scans. Both diagnostic modalities can help differentiate between a neoplastic vertebral disease, where the pathology does not usually involve intervertebral discs, and an infectious vertebral erosion, where infection invades all adjacent structures. Nevertheless, it is extremely difficult to diagnose by imaging and laboratory tests which organ has been primarily affected by the infectious process. Some cultural test can sometimes allow the isolation of infectious agents typical of the aortic or vertebral district, such as in infections caused by Treponema pallidum, opportunistic agent in immunologically compromised patients, and Mycobacterium tubercolosis. The latest can be considered the most frequent etiologic agent in vertebral erosion even if its virulence has considerably decreased in recent years. On the other hand, the widespread of imaging modalities has counterbalanced the reduced incidence of vertebral and aortic tubercular (Tbc) infections.
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Our patient presented with some peculiar features that made the etiologic diagnosis especially challenging. First, at CT scan the whole aorta presented a normal diameter apart from the site of rupture. Intraoperatively, no purulent collection was detected. Finally, all bacteriological cultures and tests were negative except for Quantiferon-TB Gold test; this test is highly sensitive for previous or current Tbc infection but is poorly specific. So our patient’s positivity to Quantiferon-TB Gold test, together with negativity to Genexpert System MTB/RIF for tuberculosis and on microbiological cultures, did not allow us to diagnose Pott’s disease. On the other hand, in their literature review, Arici et al.2 reported that positive cultures of hematoma were found only in 17.4% of patients in which those tests were performed. Historically, a vertebral disease involving the aorta has been quite always treated by surgical open drainage of the infected surroundings and aortic and vertebral open repair. Even if some authors suggest avoiding EVAR in such cases,2 we preferred to perform an endovascular aneurysm rupture exclusion because of the hemodynamic instability of the patient and the good anatomic feasibility of this low-invasive procedure. First, we planned it as a ‘‘bridging therapy’’ because of the fear for deployment of an endoprosthesis in a highly contaminated field. Moreover, we had to face the persistent supply of the periaortic hematoma by a type II endoleak, even with no decrease in hemoglobin level or hemodynamic instability persistence. In a ruptured aorta, the unending supply of the reported in up to 20% of patients.10 Fortunately, the staged spinal fixation through left lombotomy allowed a straight exploration of the involved district, so that we were able to:hematoma after EVAR can lead to a compartmental syndrome development, a complication a) b) c) d)
Drain the fluid collection and hematoma Suture the supplying lumbar arteries Collect samples for bacterial cultures Avoid the compartmental syndrome
Because we had no clear data suggesting infection, the planned open surgical conversion was
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no longer performed. At follow-up, no active infection was diagnosed and the patient considerably improved in neurologic lower limb function. Those results are probably related to the prolonged antibiotic therapy so, after 12 months, we are still strictly following him for eventual recurrence. Lumbar vertebral erosion accompanied by aortic aneurysm complete or contained rupture is a rare condition, often causing symptoms that mimic discus hernia syndrome or lower limb neuropathy; thus, potentially causing a delayed or wrong diagnosis of a life-threatening condition. EVAR can be used as an emergent bridging therapy in infectious aortic rupture or considered a more definitive treatment when no sign of infection can be detected. Surgical open exploration is sometimes needed to repair vertebral erosion and perform a radical or additional treatment. REFERENCES 1. Aydogan M, Karatoprak O, Mirzanli C, et al. Severe erosion of lumbar vertebral body because of a chronic ruptured abdominal aortic aneurysm. Spine J 2008;8:394e6. 2. Arici V, Rossi M, Antonio B, et al. Massive vertebral destruction associated with chronic rupture of infrarenal aortic aneurysm: case report and systematic review of the literature in English language. Spine (Phila Pa 1976) 2012;37: E1665e71. 3. Caynak B, Onan B, Sanisoglu I, et al. Vertebral erosion due to chronic contained rupture of an abdominal aortic aneurysm. J Vasc Surg 2008;48:1342. 4. Barros M, Lozano F, Almazan A, et al. Angio-Behc¸et with vertebral erosion: an exceptional Behc¸et’s complication and literature review. Joint Bone Spine 2004;71:577e9. 5. Loh YJ, Tay KH, Mathew S, et al. Endovascular stent graft treatment of leaking thoracic aortic tuberculous pseudoaneurysm. Singapore Med J 2007;48:e193e5. 6. Jones CS, Reilly MK, Dalsing MC, et al. Chronic contained rupture of abdominal aortic aneurysms. Arch Surg 1986;121:542e6. 7. Sterpetti AV, Blair EA, Schultz RD, et al. Sealed rupture of abdominal aortic aneurysms. J Vasc Surg 1990;11:430e5. 8. Galessiere PF, Downs AR, Greenberg HM. Chronic contained rupture of aortic aneurysms associated with vertebral erosion. Can J Surg 1994;37:23e8. 9. Booth MI, Galland RB. Chronic contained rupture of an abdominal aortic aneurysm: a case report and review of the literature. Eur J Vasc Endovasc Surg Extra 2002;3:33e5. 10. Mehta M, Kreienberg PB, Roddy SP, et al. Ruptured abdominal aortic aneurysm: endovascular program development and results. Semin Vasc Surg 2010;23:206e14.