Accepted Manuscript A contained ruptured abdominal aortic aneurysm presenting with vertebral erosion Yongqi Li, Lei Li, Dongming Zhang, Xiaomei Wang, Weidong Sun, Han Wang PII:
S0890-5096(17)30265-0
DOI:
10.1016/j.avsg.2016.09.025
Reference:
AVSG 3147
To appear in:
Annals of Vascular Surgery
Received Date: 14 April 2016 Revised Date:
7 September 2016
Accepted Date: 9 September 2016
Please cite this article as: Li Y, Li L, Zhang D, Wang X, Sun W, Wang H, A contained ruptured abdominal aortic aneurysm presenting with vertebral erosion, Annals of Vascular Surgery (2017), doi: 10.1016/j.avsg.2016.09.025. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT 1
A contained ruptured abdominal aortic aneurysm presenting with vertebral erosion
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Yongqi Li1,3, Lei Li1*, Dongming Zhang1, Xiaomei Wang2, Weidong Sun1, Han Wang1
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University, Dalian, Liaoning Province, People's Republic of China.
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Dalian, Liaoning Province, People's Republic of China.
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Ibaraki ken, Japan
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Running title: A CCR-AAA presenting with vertebral erosion
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Department of Vascular Surgery, The Second Affiliated Hospital of Dalian Medical
Department of Radiology, The Second Affiliated Hospital of Dalian Medical University,
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Current address: Laboratory of Experimental Pathology, University of Tsukuba, Tsukuba shi,
*Corresponding author: Lei Li MD
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The Second Affiliated Hospital of Dalian Medical University, 467 Zhongshan Road,
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ShaheKou District, Dalian, Liaoning, People's Republic of China.
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Email:
[email protected]/
[email protected]
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Phone: +88615541178098
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Fax: +88684672130
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ACCEPTED MANUSCRIPT Abstract
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Chronic contained rupture (CCR) of abdominal aortic aneurysm (AAA) with vertebral
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erosion is a rare condition. Although it has been reported previously, it is still liable to be
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misdiagnosed. We present a case of CCR of AAA with vertebral erosion. A brief analysis of
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similar cases reported in the last five years is presented.
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A 71-year-old male was admitted to our hospital because of severe prickling pain in his left
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thigh. Computerized tomography angiography revealed an AAA which had caused erosion of
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L3 vertebral body and the left psoas muscle. An aortotomy was performed and the excised
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aortic aneurysm replaced with a Dacron graft. Postoperative CT angiography indicated a
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normal aortic graft. The patient was discharged 13 days after the surgery. In conclusion: Pain
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in lower back and leg could be associated with vertebral erosion caused by CCR of AAA.
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Ultrasonography, CT or MRI of abdomen should be routinely performed in cases of lumbago
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that have associated risk factors for AAA.
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Keywords: abdominal aortic aneurysm, contained rupture, vertebral erosion.
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ACCEPTED MANUSCRIPT 1. Introduction
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When blood leaking from the ruptured abdominal aortic aneurysm is confined to the
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retroperitoneal cavity by the surrounding tissue, the hemodynamic parameters tend to remain
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stable, and the condition is referred to as chronic contained rupture of abdominal aortic
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aneurysm (CCR-AAA)[1]. CCR-AAA accounts for only 4% of all ruptured AAA[2].
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Occurrence of spinal erosion due to CCR-AAA is even rarer[3]. However, similar to the one
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without vertebral erosion, it is associated with a high risk of evolving into free rupture. Arici
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et al. reported a mortality rate of 17%, although comorbidities may also have contributed to
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the mortality at least in some of the cases[4]. In spite of the availability of contrast-enhanced
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computed tomography (CT) and magnetic resonance imaging to facilitate the diagnosis,
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manifestations of CCR-AAA with vertebral erosion remain ambiguous[5].
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2. Case report
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A 71-year-old male was admitted to the emergency department with complaints of severe
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prickling pain in his left thigh since the last six days. The patient had a history of smoking for
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more than 50 years (20 cigarettes per day). He was a known case of coronary heart disease,
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and had undergone coronary stent implantation in the left anterior descending artery seven
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years ago. His hemodynamic parameters were normal. Physical examination revealed a
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pulsatile mass in the abdomen. Complete blood count showed slightly decreased red blood
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cell count (3.34×1012/L) along with decreased hemoglobin (101 g/L), while the routine blood
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biochemical parameters were normal.
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CT scan of the abdomen suggested enlarged abnormal aorta, which prompted us to perform a
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ACCEPTED MANUSCRIPT computerized tomography angiography, and which revealed an abdominal aortic aneurysm
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6.4 cm in diameter, and bilateral common iliac artery aneurysms. The aortic aneurysm had
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caused erosion of the left psoas muscle and the anterior 30% of the L3 vertebral body (Figure
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1 & 2). In addition, a small amount of contrast medium was observed in the hematoma
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surrounding the aorta, which suggested leakage of blood from the abdominal aorta, which
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was confined in the pseudoaneurysm. Based on the clinical features, laboratory results and
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imaging findings, the patient was diagnosed as a case of CCR of AAA.
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An open abdomen surgery was performed for the repair of the aortic aneurysm. After cross
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clamping the abdominal aorta, aortotomy was performed. A defect in the posterior wall of
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aorta along with perforation in the vertebral body, measuring 3 × 4 cm, was found. The
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vertebral body was filled with bone wax and the excised aortic aneurysm replaced with an 8
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mm × 16 mm Y-shaped Dacron graft. Pathological examination of the excised arterial wall
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did not reveal any special findings. The patient developed acute left heart failure shortly after
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the surgery, which was resolved with medication. Repeat CT angiography demonstrated a
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normal aortic graft and no postoperative complications occurred (Figure 3). Thirteen days
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after the surgery, the patient was discharged; pain in his left extremity was relieved.
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3. Review of Literature and Discussion
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Ever since the first case of CCR-AAA causing lumbar vertebral erosion was reported by Katz
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et al[6] in 1962, less than 100 similar cases have been documented worldwide. A case of
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CCR-AAA is liable to be mistaken for a vertebral or retroperitoneal tumor, psoas or spinal
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abscess, spondylitis or osteoporosis, owing to its rarity, lack of typical manifestations,
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ACCEPTED MANUSCRIPT findings of vertebral erosion, and the presence of low density mass surrounding the
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abdominal aorta in the retroperitoneal space on CT scan. In order to characterize the clinical
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features of this disease, we searched the PubMed database for reports on CCR-AAA that
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caused lumbar vertebral erosion, published since 2011. We retrieved 12 reports (Table 1). A
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brief analysis of these cases along with findings of the present case report is presented.
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The pathogenesis of AAA is still obscure. Smoking, old age, male gender, atherosclerotic
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arterial disease, dyslipidemia, family history are common risk factors associated with
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AAA[7]. In addition, infection and inflammation are also reported to be involved in its
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pathogenesis[8]. Since our patient did not present with a fever, nor manifested any abnormal
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biochemical or hematological parameters suggestive of infection and inflammation, the
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possibility of infection and vasculitis was deemed highly unlikely Among the 12 cases
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reported above, all were males except one. Average age of the patients was 62.25 years; 10
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cases (83.3%) were > 50 years of age. Of these, four patients had a history of smoking,
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dyslipidemia, hypertension, or diabetes. Two cases were diagnosed with Behcet’s disease, an
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autoimmune disease which damages various organs, including vascular system[9, 10]. One
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patient was a case of osteogenesis imperfecta who had a six-year-long history of back pain
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[11]. One patient had a history of chronic renal failure and emphysema, and had undergone
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coronary artery bypass grafting (CABG). This patient died of pneumonia during
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treatment[12].
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Most patients complained of discomfort that ranged from weeks to months. The most
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common symptoms of CCR-AAA with vertebral erosion were lower back pain (N=10;
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83.3%), followed by leg pain or claudication (N=6; 50%), pulsatile mass (N=4; 33.3%),
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ACCEPTED MANUSCRIPT abdominal pain (N=1; 8.3%), shoulder pain (N=1; 8.3%), and hematemesis (N=1; 8.3%). One
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patient who presented with sudden onset of hematemesis was found to have aortoenteric
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fistula, a rare complication of AAA[13].
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Lower back pain is a symptom with low specificity, although it was the most common
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symptom of CCR-AAA with vertebral erosion. In the U.S.A, lower back pain is the
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fifth-leading cause of hospital admissions, and the third most common cause of surgical
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procedures[14]. In order to make the right diagnosis of CCR-AAA with vertebral erosion, it is
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strongly suggested that ultrasound scan, CT or MRI of abdomen be performed routinely in a
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patient with risk factors associated with AAA and presenting with lumbago.
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In the present case, the patient complained of severe prickling pain in the left thigh, which, in
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all probability, was caused by compression of the left femoral nerve by the enlarged
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pseudoaneurysm. Femoral nerve arises from divisions of L2-L4 lumbar nerves and provides
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sensory innervations over the front and inner sides of thigh. It descends through the psoas
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major muscle, and eventually runs into thigh. Although it was only six days since the advent
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of the presenting symptom, it is possible that the CCR-AAA with eroded vertebrae had
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existed for a much longer time. Perhaps only when the CCR-AAA began to compress the left
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femoral nerve, did the pricking pain start.
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CCR-AAA may cause erosion of any lumbar vertebra. The most commonly involved
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vertebrae in previous reports were (in that order): L3 (N=10; 90.9%), L4 (N=6; 54.5%), and
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L2 (N=4; 36.4%). In the present case the enlarged pseudoaneurysm had eroded the anterior
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30% of the third lumbar vertebral body, and had formed a cavity measuring 3 cm × 4 cm in
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size. The pathogenesis of vertebral erosion in CCR-AAA is not clear. Some researchers
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ACCEPTED MANUSCRIPT believe that repetitive beats from pulsating aorta and blood jet leads to the destruction of the
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vertebra, while others have attributed it to infection and/or inflammation[4]. However, no
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correlation has been reported between the diameter of CCR-AAA and vertebral erosion.
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There are two main surgical methods for treating CCR-AAA: open repair of the aortic
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aneurysm and endovascular aneurysm repair (EVAR). The choice of surgical approach is
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based on the parameters of the ruptured aneurysm, the patient’s general condition,
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institutional capacity, and on surgeon’s preferences [7]. In the present case, we opted for open
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abdomen repair instead of EVAR as the aneurysmal neck was not long enough to
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accommodate a stent. The decision to perform surgical reconstruction of the eroded vertebrae
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is a crucial consideration in these patients as well. The site and extent of the erosion, along
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with the patient’s general condition are key factors that influence treatment strategy[15]. We
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did not perform spinal fixation on the eroded vertebra immediately in the present case
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because the patients’ spine was considered stable enough based on the three-column theory.
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Follow-up examination also showed that the erosion did not affect his normal life.
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4. Conclusion
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CCR-AAA with vertebral erosion is a rare but potentially life threatening condition which
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usually presents with pain in lower back and leg. The condition could be associated with
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erosion of any lumbar vertebral body, but L3 and L4 vertebral bodies are most commonly
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involved. It is strongly recommended that ultrasonography, CT or MRI of abdomen be
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performed routinely in cases of lumbago who manifest risk factors for AAA.
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ACCEPTED MANUSCRIPT Figure legends
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Figure 1: Horizontal plane of computerized tomography angiography showing eroded
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vertebral body and left psoas muscle due to enlarged psudoaneurysm.
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Figure 2: Sagittal plane of computerized tomography angiography showing the eroded third
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lumbar vertebral body
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Figure 3: Postoperative computerized tomography angiography of the abdominal aorta
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Table 1: Basic demographic and clinical information of patients in 23 reports Main symptoms and
Treatment Treatment of Eroded
Diameter Previous
their duration and Sex
vertebrae of AAA
Low back pain, 63 M
claudication, pulsatile mass. 4m
Jimenez-Viseu Pinheiro et 75 F
Low back pain, pulsatile
L4
4 cm
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Alshafei et al. [16]
L2-L4 mass. 6m
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al. [15]
of AAA
spine
Smoking,
Open
No surgical
dyslipidemia,
Surgery
treatment
55 M
L3-L4
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pain. 2y
Smoking,
Discharged dyslipidemia
68 M
treatment
Stabilization
EVAR
No surgical
Behcet’s disease
Discharged treatment
Low back pain, Singla et al. [17]
No surgical Spinal
5 cm
5 cm
EVAR L3
5.5 cm
Discharged treatment
Low back pain, 38 M
Behcet’s disease Open L3
62 M
Low back and leg pain
No surgical
4 cm
claudication in right leg Nakano et al. [18]
No surgical
Diabetes
pulsatile mass. 4m
Kara et al. [9]
Discharged
hypertension
Shoulder pain, low back Örücü et al. [10]
Outcome
history
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(m =month; y= year)
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Age (y) Authors
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L4
Not 12
Discharged 6 years ago
surgery
treatment
Not described
Open
Lumbar
Discharged
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described
surgery
interbody
Acute ischemia of left leg, abdominal pain with Stefanczyk et al. [19]
59 M
L3-L4
73 M
Back pain. 6y
L2-L3
Low back pain. 6m
L2-L3
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Arici et al. [4]
68 M
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Davies et al. [11]
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region for weeks
No surgical
Not described
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radiation to the lumbar
9.5 cm
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fusion
Osteogenesis
Discharged EVAR
treatment
Not
Not described
14 cm
10.4 cm
Discharged imperfecta
described Open
Spinal
surgery
stabilization
Hypertension
Discharged
Chronic renal
No surgical No surgical
failure,
treatment
treatment
83 M
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Left anterolateral thigh Keller et al. [12]
L2-L3
5 cm
underwent
Death
and knee pain. 3d
Tsubota et al. [20]
60 M
CABG, emphysema
Right thigh and right knee L3-L4 13
11 cm
Not described
Open
No surgical
Discharged
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pain, low back pain,
surgery
treatment
Open
No surgical
surgery
treatment
Not
Not
hematemesis
described
described history
43 M
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CABG, Coronary artery bypass graft
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Sudden onset of
No positive
SC
Yie et al. [13]
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pulsatile mass. 2m
14
Discharged
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