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ORIGINAL ARTICLE
Management of true visceral artery aneurysms in 31 cases S. Regus ∗, W. Lang Department of Vascular Surgery, University Hospital, Krankenhausstrasse 12, 91054 Erlangen, Germany
KEYWORDS Visceral artery aneurysm; False aneurysm; Coil embolization; Indications; Chronic pancreatitis
∗
Summary Introduction: True visceral artery aneurysms (VAA) should be treated under elective conditions in dependency on maximum diameter. In this respect, the traditional accepted threshold is 2 cm, whereas VAA sizing less than 2 cm should conservatively be observed without invasive treatment. The aim of this study was to review differences in the treatment outcome over three decades. Material and methods: This was a retrospective review of all treated VAAs at one institution from 1985 to 2015. Patients demographics, aneurysm characteristics, management and outcome were recorded with special regard to differences in the course of time. Results: Thirty-one true VAA in 29 patients (74% female) were repaired (5 ruptured, 26 intact). Mean diameter was 30.27 ± 11 mm for intact and 38.0 ± 8.5 mm for ruptured VAA (rVAA) (P = NS). Most patients were asymptomatic (67.8% asymptomatic, 16.1% symptomatic without rupture and 16.1% with rupture). There was a vice-versa situation in chosen treatment techniques between the first (1985—2000) and the second (2001—2015) time period [first period: 75% open repair (OR) and 25% endovascular repair (ER); second period: 27% OR and 73% ER; P = 0.009]. OR included aneurysm ligation and resection with (end-to-end-anastomosis, graft interposition or without blood flow reconstruction), while ER was exclusively coil embolization with sacrifice of all parent afferent and efferent arteries. Immediate technical success was 81% for all procedures. There was a trend toward higher technical success rate of VAA being treated in second time period, but we found no significant differences (69% in the first, 93% in the second; P = 0.101). Conversion to OR due to technical failures was necessary after 3 endovascular repairs (20%). The overall 30-day-mortality rate decreases in the course of time (25% in the first and 0% in the second period; P = 0.038). Furthermore, there was a lower 30-day mortaliy rate after ER of all VAA (elective and urgent repair) (20% after OR, 0% after ER; P = 0.038). There was no decrease in 30-day mortality rate of rVAA (100% in the first and 20% in the second period; P = NS).
Corresponding author. E-mail address:
[email protected] (S. Regus).
http://dx.doi.org/10.1016/j.jviscsurg.2016.03.008 1878-7886/© 2016 Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Regus S, Lang W. Management of true visceral artery aneurysms in 31 cases. Journal of Visceral Surgery (2016), http://dx.doi.org/10.1016/j.jviscsurg.2016.03.008
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S. Regus, W. Lang Conclusion: In the fact of medical progress and a growing number of endovascular procedures, this study presents a decrease in mortality rate after elective aneurysm repair over three decades. This might become an argument to reduce the 2-cm threshold in highly selected individuals. © 2016 Elsevier Masson SAS. All rights reserved.
Introduction Visceral artery aneurysms (VAA) are a rare disease with an estimated incidence about 0.1 to 2.0% [1]. Nowadays advanced imaging techniques as computed tomography or magnetic resonance angiography are widely used and therefore incidental detection of VAA increased over the years [2]. Rupture is the most serious complication of VAA and invasive management primarily should avoid this life-threatening condition. Asymptomatic true VAA outside childbearing age or pregnancy should be treated if maximum diameter is 2 cm or more [3,4]. Theses traditional recommendations to exclude VAA are mainly dated from a period before endovascular techniques were available and open surgery was the only way to exclude aneurysms [5]. Since the early 20th century endovascular management in different techniques has increasingly been used with promising results especially in multimorbid poor surgical candidates [6]. Furthermore medical progress, especially the management of critical illness as hemorrhagic shock, has evolved over the last decades [7]. The aim of this study was to analyze early and follow-up results after elective treatment of VAA with special regard to differences in the course of time and therefore possible effects on future indications of invasive treatment.
the afferent artery. Furthermore, in cases of persisting blood flow, coils were packed into the aneurysm sac. A sacrifice of all parent afferent and efferent arteries was done. There were no other endovascular techniques employed (i.e. stent placement or embolization using liquid agents) and no use of minimal invasive laparoscopic treatment in this study.
Definitions Definitions were as follows: technical success (no further blood flow within the aneurysm sac), conversion to open surgery (technical failure followed by surgical repair), major complications (stroke, myocardial infarction, bleeding leading to surgery or death). Hemodynamic shock was defined as a shock-index > 1 (heart rate divided by systolic blood pressure) when admitting the hospital.
Diagnostic work-up In every elective repaired VAA, the preoperative diagnostic tool included duplex ultrasound (DUS), computed tomographic angiography (CTA) or magnetic resonance angiography (MRA), whereas 3 of the 5 patients with ruptured VAA (rVAA) underwent emergency surgery without vascular imaging.
Material and methods
Follow-up program
Patients
Follow-up examinations included DUS two times a year for the first 24-month, afterwards in case of inconspicuous findings in annual intervals. CTA or MRA was carried out in cases where DUS was technically not sufficient (e.g. in the presence of obesity) or in the suspicion of reperfusion due to technical failures.
Thirty-one true VAA in 29 patients were repaired in our institution from January 1985 to June 2015 by open or endovascular techniques. To analyze differences in the course of time, patients were divided into two groups: 16 VAA (group 1) were treated from January 1985 to December 2000 and 15 VAA (group 2) from January 2001 to June 2015. Patients under surveillance were excluded as well as false VAA due to their different indications and etiology.
Indications and treatment techniques Indications for open repair (OR) or endovascular repair (ER) was a diameter of 2 cm or more, every VAA in women of childbearing age and every symptomatic or ruptured VAA (rVAA) without regard to size. OR was performed by aneurysm resection or ligation with (end-toend-anastomosis, vein-interposition or bypass grafting) or without (ligation or resection) blood flow reconstruction. All endovascular interventions were performed in an angiosuite by interventional radiologists, under local anaesthesia, supplemented with sedation or analgesia when required. An unilateral femoral approach was preferentially used and all patients underwent intravenous administration of 5000 IU of sodium heparin at the time of arterial catheterization. In detail, coils were packed into the efferent and afterwards
Data collection and analysis Data collection was done in a retrospective manner by analyzing medical records. Statistical analysis was performed by means of SPSS 21.0 for Windows (SPSS Inc., Chicago, Illinois). Differences between groups were analyzed for categorical variables (Chi2 ) and for numerous (t-test). The ˛ level for significant tests was 0.05. A P > 0.05 represents no significant (NS) differences.
Results Patients Demographic data and comorbid conditions are shown in Table 1. There was no significant difference between the two groups. One asymptomatic female patient in group 1 received OR during pregnancy. Five patients (16%) suffered from hemorrhagic shock due to rupture and emergency
Please cite this article in press as: Regus S, Lang W. Management of true visceral artery aneurysms in 31 cases. Journal of Visceral Surgery (2016), http://dx.doi.org/10.1016/j.jviscsurg.2016.03.008
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Visceral artery aneurysms in the course of time Table 1
3
Demographic data and comorbidities. All (n = 31)
Age
Gender (female) Hypertension Diabetes mellitus Hyperuricemia Hyperlipidemia Nicotine abuse
1985—2000 (n = 16)
P*
2001—2015 (n = 15)
Mean ± SD
Range
Mean ± SD
Range
Mean ± SD
Range
54.8 ± 11.5
(24—86)
58.4 ± 10.8
(39—86)
50.9 ± 14.6
(24—70)
n
%
n
%
n
%
23 17 10 6 14 13
74 55 32 19 45 42
13 11 5 3 10 9
81 69 31 19 63 56
10 6 5 3 4 4
67 40 33 20 27 25
0.112
0.354 0.106 0.602 0.641 0.073 0.096
SD: standard deviation; n: numbers; %: percent. * When comparing the two time periods.
repair was necessary [2 (12%) in group 1 and 3 (20%) in group 2, P = NS].
Aneurysms and treatment details Aneurysm data, treatment details and follow-up outcome of all VAA and comparing the two groups are shown in Table 2.
Table 2
Mean diameter of intact VAA was 30.27 ± 11 mm and 38.00 ± 8.5 mm for ruptured VAA (P = NS). The most common etiology for aneurysmal disease was atherosclerosis in 22 cases (71%), followed by collagen dysplasia in 3 (9.6%), high flow situation in collateral pathways due to mesenteric or celiac stenosis in 3 (9.6%) and unknown etiology in 3 cases (9.6%).
Aneurysm data, treatment details and follow-up. All (n = 31)
Symptoms Asymptomatic Symptomatic Ruptured
Diameter (millimeters)
Location Splenic Hepatic Celiac Pancreaticoduodenal Technique Open surgery Coil embolization 30-day mortality Morbidity Success 30-day-reintervention Hospital stay (days)
Follow- up (month)
1985—2000 (n = 16)
P*
2001—2015 (n = 15)
n
%
n
%
n
%
20 6 5
65 19 16
10 4 2
63 25 12
10 2 3
0.812 67 13 20
Mean ± SD
Range
Mean ± SD
Range
Mean ± SD
Range
31.5 ± 10.9
(20—65)
32.8 ± 12.2
(20—65)
30.1 ± 9.7
(20—54)
n
%
n
%
n
%
20 7 2 2
65 22 6.5 6.5
12 3 1 —
75 19 6 —
8 4 1 2
53 27 7 13
16 15 4 10 25 3 8.35 ± 7.0
52 48 13 32 81 10 (1—34)
12 4 4 9 11 2 11.2 ± 8.0
75 25 25 56 69 12 (1—34)
4 11 0 1 14 1 4.5 ± 3.4
27 73 0 7 93 7 (2—13)
Mean ± SD
Range
Mean ± SD
Range
Mean ± SD
Range
27.13 ± 21.2
(0—73)
26.9 ± 21.2
(0—73)
29.3 ± 20.2
(5—68)
0.613
0.405 0.525 0.797 0.627 0.009
0.038 0.047 0.101 0.583 0.006
0.936
n: number; %: percent; SD: standard deviation. * When comparing the two time periods.
Please cite this article in press as: Regus S, Lang W. Management of true visceral artery aneurysms in 31 cases. Journal of Visceral Surgery (2016), http://dx.doi.org/10.1016/j.jviscsurg.2016.03.008
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S. Regus, W. Lang Table 3
Outcome after elective open or endovascular repair of 26 visceral artery aneurysms (VAA). All (n = 26)
Success 30-day-reintervention Morbidity 30-day mortality
Hospital stay (days) Follow-up (month)
OR (n = 11)
P*
ER (n = 15)
n
%
n
%
n
%
21 3 6 1
81 12 23 4
9 0 3 1
82 0 27 9
12 3 3 0
80 20 20 0
Mean ± SD
Range
Mean ± SD
Range
Mean ± SD
Range
9±7 27.34 ± 21.5
(1—34) (0—73)
13 ± 9 26.09 ± 23.8
(1—34) (0—73)
5±4 34.9 ± 17.7
(2—14) (5—68)
0.907 0.238 0.176 0.423
0.006 0.326
OR: open repair; ER: endovascular repair; n: number; %: percent; SD: standard deviation. * When comparing OR and ER.
In particular, OR was done with reconstruction of blood flow in 9 (56%) and without in 7 cases (44%) reconstruction. OR was the preferred treatment technique from 1985 to 2000, while ER was mainly used from 2001 to 2015 (Table 2).
died 13 month and the other 18 month after aneurysm repair), 3 patients were lost. Finally, we compared the outcome after OR and ER without respect to the time period. We excluded the 5 rVAA for better analyzing the outcome after elective treatment. The results are shown in Table 3.
Outcomes Immediate technical success was achieved in 25 cases (80.6%). Six procedures (19.4%) failed, three after OR and three after ER. The failed surgical procedures were due to hemorrhagic shock after rupture and patients death, whereas endovascular failures were associated with endoleaks and the necessity for early intervention (30-day reintervention rate 9.7%). Conversion to OR within 30 days was necessary in 3 cases (20%). The major complication rate was 32% (10 cases), in detail 4 bleedings leading to surgery, 4 in-hospital death and 2 endoleaks. In detail, the morbidity after ER was 20% (2 endoleaks and 1 bleeding leading to surgery) and 44% after OR (4 death and 3 bleeding leading to surgery), but these results were not significant (P = 0.231). Splenic infarction after ER or OR without reconstruction of splenic blood flow occurred in 6 cases (31%) (3 cases/28% after OR and 3 cases/33% after ER, P = NS). Patients suffered from mild abdominal pain, which disappeared without the need for further invasive treatment. The 30-day mortality rate was 20% following OR and 0% after ER (P = 0.038). Four patients died during hospital stay. All in-hospital deaths were documented in symptomatic patients, most of all (75%) in ruptured aneurysms. Two patients were female; distribution of ruptured VAA with deathly outcome was splenic artery (2) and celiac artery (2). All patients who died after aneurysm repair received OR (two with and two without blood flow reconstruction). Five patients (16%) admitted hospital with hemodynamic shock after aneurysm rupture (1 splenic, 2 celiac, 1 hepatic, 1 pancreaticoduodenal). OR was performed in all of these 5 cases, in 2 cases without and in 3 cases with arterial reconstruction. The 30-day mortality rate for rVAA was 60% (2 patients died in operating theatre, 1 patient on the first day after surgical repair). In the first time period, the 30-day mortality after rupture was 100% (2 patients died), while in the second period from 2001 to 2015, it was 20% (1 of 3 patients died, P = NS). During follow-up, 2 patients died nonaneurysm related due to myocardial infarction (1 patient
Discussion In this study, we found a significant reduced morbidity and mortality in the second time period from 2001 to 2015. This could be associated with the observation of higher mortality after OR, which was the preferred technique in the first time period. Furthermore, we observed a reduced morbidity after ER, but these results were not significant due to the low number of patients. Otherwise there could be a selection bias because rVAA were exclusively treated by OR. This is supported by the fact that we found no significant differences in immediate outcome results after OR and ER if ruptured aneurysms were excluded. Recommendations for treatment of VAA are aimed at preventing potentially life-threatening rupture. The risk of rupture depends on location, type and etiology and is reported to be up to 20% [2]. In the presence of rupture with hemorrhagic shock, the associated mortality rate is very high about 70—100% [8—10]. Otherwise elective repair represents a success rate up to 100% and a perioperative mortality rate of 3% [11]. Recently published data confirm the excellent technical success rates of elective repaired VAA with no perioperative death [12]. Table 4 provides an overview about previously published data about open and endovascular management of visceral aneurysms. Despite great medical advancement over the last decades, especially development of less invasive endovascular techniques and excellent shock management, there seems to be no decrease in mortality rate after invasive management of rVAA [5,13]. Our data confirm these findings in detail, there was a 30-day mortality rate of 60% with no significant difference in the course of time.
Elective repair Due to high risk for rupture regardless of maximum diameter, every false VAA and all true VAA during pregnancy or
Please cite this article in press as: Regus S, Lang W. Management of true visceral artery aneurysms in 31 cases. Journal of Visceral Surgery (2016), http://dx.doi.org/10.1016/j.jviscsurg.2016.03.008
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Visceral artery aneurysms in the course of time Table 4
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Previously published data about open and endovascular repair of true and false visceral artery aneurysms.
Author
Year
Time period
n
(r)
Technical success (%)
Complications
30-day mortality
Follow-up
Shukla et al. [15] Spiliopoulo Marone Carmeci Chiesa et al. [16] Ferrero Saltzberg Tulsyan et al. [1] Dorigo et al. [12]
2015
2003—2013
181
(77)
98
13.7% (r)
1.0% (int)
13.0% (r)
0% (int)
26 months
2011 2011 2000 2005
2000—2010 1988—2010 1980—1998 1988—2002
21 94 31 31
(2) (0) (19) (3)
100 95 85 57.1
5% 9.4% 0% 7.1% (O)
14.3% (E)
3% 1.3% 3.2% 3.6% (O)
0% (E)
19 months 42 months NR 68 months
2011 2005 2007
2000—2010 1990—2009 1997—2005
32 27 48
(6) (4) (5)
85 94.4 98
9% 22.2% 2%
9.4% 0% 8.3%
34 months 14.9 months NR
2015
2007—2013
26
(0)
89
12%
0%
18 months
r: ruptured; int: intact; n, number.
in women of childbearing age should be repaired [13]. Furthermore, renal artery aneurysm should be treated toward a smaller diameter up to 1.5 cm because of higher mortality rate and the risk of renal loss in case of rupture [14]. True VAA located in splenic, hepatic, celiac and pancreatiduodenal area outside pregnancy or childbearing age should traditionally be excluded in an elective setting if sizing 2 cm or more [15]. Previously published data recommend a more aggressive and timely treatment of VAA because of low mortality rates in case of elective repair [16]. Our data confirm these recommendations showing a significant reduced morbidity and mortality rate after elective VAA repair in the course of time, whereas there was no significant improvement in survival after emergency surgery. The better outcome of elective repair in the course of time could be due to nowadays the widespread use of computed tomographic angiography and therefore diagnosing in an early stage with smaller diameters. Indeed, there was no significant difference in aneurysm data, especially location and size, or comorbid conditions between the early (treated from 1985 to 2000) and the later group (2001 to 2015). Therefore, we do not believe differences in mortality rates are caused by selection or performance bias as treating fitter patients with smaller aneurysms in the course of time. In contrast, as a result of the changing demographic structure it may be expected that patients will be treated even in advanced age with a higher morbidity. Decrease in mortality rate after elective repair in the study presented could be caused by a significant increased number of interventional treatment in the years from 2001 to 2015 compared to the time period from 1985 to 2000. This fact seems for us to be the main reason for the significant shorter hospital stay in the second time period. In case of ER, we unexceptionally performed coil embolization with packing coils into the efferent and afferent artery, finally if necessary into the aneurysm sac. This is the standard procedure with the best expertise at our institution. Even if the rate of splenic infarction was about 30%, there was no need for invasive repair and patients were asymptomatic after conservative medical treatment. Promising data about flow-diverting stents for the treatment of VAA has been published and could prevent end-organ infarction and therefore reduce the already low morbidity
rates [17]. We have no experience in these new techniques. Furthermore up to now, we did not use the laparoscopic approach in our institution, even if it has been reported to be a promising and less invasive procedure [18,19]. Nowadays due to widespread available advanced imaging techniques, most VAA are incidental findings [20]. After diagnosis has been made, many patients inform themselves about their disease via the Internet. Furthermore patients have a high expectation to medical services and the highest life expectancy than ever before. A significant number of patients, especially when being fit and healthy, are often afraid of aneurysm rupture and demand early performance of invasive treatment. For those patients, a more aggressive treatment management could be favourable even if maximum diameter is less than 2 cm.
Limitations Limitation of the study presented is the retrospective design and the risk of reporting as well as performance bias. Furthermore considering the small number of rVAA, we cannot reach significant differences in perioperative mortality rate in the course of time. There is a trend to better outcome in the second time period, but this does not indicate a reduction of mortality rates of rVAA in the course of the last 30 years. Our data are rather comparable with previously published data [21] and we believe, in spite of medical progress, the high mortality rate of rVAA will remain unchanged.
Conclusion Traditional recommendations to treat asymptomatic true VAA are focused on a size more than 2 cm, but they ignore currently improved outcome of elective repair compared to unchanged high mortality rates associated with aneurysm rupture. We believe in individual cases the threshold of maximum diameter should be amended toward less than 2 cm.
Disclosure of interest The authors declare that they have no competing interest.
Please cite this article in press as: Regus S, Lang W. Management of true visceral artery aneurysms in 31 cases. Journal of Visceral Surgery (2016), http://dx.doi.org/10.1016/j.jviscsurg.2016.03.008
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Please cite this article in press as: Regus S, Lang W. Management of true visceral artery aneurysms in 31 cases. Journal of Visceral Surgery (2016), http://dx.doi.org/10.1016/j.jviscsurg.2016.03.008