A systematic review on clinical features and management of true giant splenic artery aneurysms

A systematic review on clinical features and management of true giant splenic artery aneurysms Hytham K. S. Hamid, MRCSEd,a Abd Elaziz A. Suliman, MD, FRCS,a Gabriele Piffaretti, MD,b Stavros Spiliopoulos, MD,c Raphael Tetreau, MD,d Matteo Tozzi, MD,b and Raffaelle Pulli, MD,e Khartoum, Sudan; Varese and Bari, Italy; Rion, Greece; and Montpellier, France

ABSTRACT Background: True giant splenic artery aneurysms (GSAAs) >5 cm are rare and present unique therapeutic challenges. The aim of this study was to evaluate the anatomic and clinical characteristics of these lesions and the current surgical and endovascular techniques available for their treatment. Methods: A systematic review of the literature from 2004 to 2018 and the personal experience of the authors with management of GSAAs are presented. A total of 92 GSAA cases were reviewed. Analyses were performed on anatomic and clinical features and management modalities and outcomes of GSAA, including reintervention, morbidity, and mortality. Results: GSAA presented at a mean age of 56.1 6 17.3 years, with no sex predilection; 73% were symptomatic at presentation. Abdominal pain was the presenting symptom in >50% of cases; 34% percent were ruptured, with an overall mortality rate of 12.5%. This group often presented with gastrointestinal bleeding or hemodynamic collapse. The aneurysms were almost evenly distributed across the splenic artery and were not uncommonly associated with arteriovenous fistula formation (8.7%). There were 88 patients who had surgical (53.4%), endovascular (44.3%), or combination (2.3%) therapy. The most commonly performed procedure was aneurysmectomy and splenectomy with or without additional resection. Overall, surgical treatment had a lower morbidity (P ¼ .041) than endovascular therapy and comparable reintervention and mortality rates. Conclusions: GSAAs are uncommon vascular lesions, with distinct clinical features and aneurysm characteristics. Considering their high risk of rupture, timely diagnosis and management are essential to attain a satisfactory outcome. Surgery remains the standard treatment of these lesions. Endovascular intervention is a viable alternative in high-risk patients, particularly those with lesions <10 cm or with anomalous origin. (J Vasc Surg 2019;-:1-10.) Keywords: Giant; Splenic; Aneurysms; Endovascular; Surgical; Management

Splenic artery aneurysms (SAAs) are being increasingly diagnosed by virtue of the advances made in the realm of radiologic studies. The estimated incidence rate of SAAs runs between 0.098% and 10.4% in autopsy studies.1,2 In one of the largest contemporary series, the mean diameter of these lesions was 3.1 cm, whereas the maximum diameter did not exceed 5 cm.3 True giant SAAs (GSAAs) >5 cm are uncommon. The relevance of recognizing these clinical entities is mainly to avoid

rupture with otherwise lethal exsanguination. Moreover, the huge size of these lesions and the associated complex anatomy render the surgical and endovascular management difficult and highly demanding.4 This study reviewed the literature and presents the clinical features and management outcomes of 92 cases of GSAAs >5 cm.

METHODS From the Vascular Surgery Division, Department of Surgery, Soba University a

Hospital, Khartoum ; the Department of Medicine and Surgery, University of Insubria School of Medicine, Vareseb; the Department of Diagnostic and Interventional Radiology, Patras University Hospital, Rionc; Centre d’Imagerie Médicale, Institut du Cancer, Montpellierd; and the Vascular and Endovascular Surgery Unit, University of Bari, Bari.e Author conflict of interest: none. Additional material for this article may be found online at www.jvascsurg.org. Correspondence: Hytham K.S. Hamid, MRCSEd, Vascular Surgery Division, Department of Surgery, Soba University Hospital, Madani Rd, Khartoum, Sudan (e-mail: [email protected]). The editors and reviewers of this article have no relevant financial relationships to disclose per the JVS policy that requires reviewers to decline review of any manuscript for which they may have a conflict of interest. 0741-5214 Copyright Ó 2019 by the Society for Vascular Surgery. Published by Elsevier Inc. https://doi.org/10.1016/j.jvs.2019.09.026

A systematic search in the English literature was performed to identify publications on GSAA (defined as true SAA >5 cm in maximum diameter) during the period between January 2004 and December 2018 through two search strategies. The initial search strategy involved searches through the PubMed databases using the keywords “giant,” “large,” “huge,” “splenic artery,” “visceral artery,” and “aneurysm” in various combinations. Details of search methodology and number of articles can be found in the Supplementary Fig (online only). The PubMed citations were then used to obtain details of the reported cases. The second strategy involved review of the references for further relevant articles on GSAA cited by relevant publications identified through the initial strategy. 1

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All articles with original data on characteristics and treatment outcomes of true SAAs were included in the initial search. Exclusion criteria included articles describing pseudoaneurysms, articles not describing true SAA >5 cm, and articles with no clear distinction between true SAA and pseudoaneurysm. Articles published by the same authors were carefully screened to identify duplicate cases. Overall, 75 publications with adequate details met the eligibility criteria.4-78 Another six publications provided inadequate details about clinical features or management of GSAA and were excluded; most were case reports describing one case (Supplementary References, online only). Two unreported cases from our experience were also included. The total number of cases reviewed was 92. The majority of the reported cases had originated from Asia (n ¼ 41 [44.6%]), followed by Europe (n ¼ 37 [40.2%]) and North America (n ¼ 9 [9.8%]). Three cases originated from Latin America (n ¼ 3 [3.2%]) and two from Africa (n ¼ 2 [2.2%]). All available publications were carefully analyzed. The corresponding authors were contacted by e-mail when required. Clinical data were collected on demographics, aneurysm characteristics, clinical presentations, imaging investigations, primary treatment details (open, laparoscopic, endovascular, and conservative), complications, reintervention, follow-up, and mortality.

STATISTICAL ANALYSIS Continuous data were summarized as means and standard deviations, if possible, and categorical data were summarized using numbers and percentages. Continuous variables were compared between groups using independent Student t-test. The association between categorical variables was assessed using Pearson c2 test and Fisher exact test as appropriate. Considering the difference in management strategies, patients were dichotomized into two groups based on the size of the aneurysm (#10 cm and >10 cm). A two-sided significance level of P < .05 was considered to be statistically significant. Statistical analysis was performed using SPSS software version 22 for Windows (IBM Corp, Armonk, NY).

RESULTS Forty-seven (51.1%) patients were female, with a mean age of 56.1 6 17.3 (range, 20-90 years). The aneurysms had a mean maximum diameter of 8.6 6 3.4 cm (range, 5.1-21 cm). Table I summarizes clinical features, causes, and anatomic characteristics of the GSAA. The most common reported cause of GSAA was essential hypertension (24%). For GSAA #10 cm, other common causes included pregnancy and portal hypertension. Thirty-two (34.8%) were ruptured, and the site of rupture was described for 31 patients: the peritoneal cavity (32.2%), retroperitoneum (22.6%), gastrointestinal tract (22.6%), vein (16.1%), and multiple sites (6.5%).

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Sixty-seven patients (72.8%) were symptomatic; symptoms were directly related to aneurysm in 86.6%. Abdominal pain was the most common presenting symptom, occurring in 47 (51.1%) patients; 33 (35.8%) had bleeding. Of this group, 18 (54.5%) were hemodynamically unstable. The bleeding was directly related to aneurysm in 27 patients. In another six patients, the bleeding was not directly related to aneurysm, and this included primary and secondary variceal bleeding, ruptured left gastric artery aneurysm, and gastrointestinal bleeding of unknown origin. Overall, GSAAs were almost equally distributed between the three segments of the splenic artery, except for GSAAs >10 cm, in which the middle third was the most common site (56.5%). In the ruptured group, however, 80.6% were located in the middle and distal artery, and the mortality rate was 12.5%. Eight patients (8.7%) had multiple SAAs. Another five patients (5.4%) had one or more other visceral aneurysmsdleft gastric, right gastroepiploic, superior mesenteric, hepatic, and celiac aneurysm. GSAA developed in splenic arteries with anomalous origin from the superior mesenteric or hepatic artery in six (6.5%) patients, and eight (8.7%) had arteriovenous fistula with either the portal or splenic vein. The morphologic appearance was described for 74 GSAAs; most (92%) were saccular. Conventional computed tomography (CT) was the most commonly performed diagnostic study (70.7%), followed by conventional or Doppler ultrasound (47.8%), angiography (40.2%), and CT angiography (39.1%). Magnetic resonance imaging (4.3%), plain radiography (3.3%), and contrast-enhanced ultrasound (1.1%) were rarely used. Endoscopy was reportedly diagnostic in one (1.1%) patient. Finally, immediate laparotomy or autopsy was diagnostic in five patients (5.4%).

MANAGEMENT Overall, of the 88 patients who had intervention, 47 (53.4%) were initially treated with primary surgery, 39 (44.3%) with endovascular therapy, and 2 (2.3%) with combined therapy. The Fig demonstrates the number of cases treated with primary surgery or endovascular therapy during the study period. One elderly frail patient was managed expectantly without complications for a period of 12 months. Three patients died of aneurysm rupture before any treatment.

PRIMARY SURGERY The surgical procedures are listed in Table II. Surgical intervention was performed in 47 patients with a success rate of 97.9%. Open surgical treatment included aneurysmectomy with splenectomy (30/47), ligation with or without splenectomy (8/47), and aneurysmectomy with vascular reconstruction (7/47). Two other patients had laparoscopic aneurysmectomy with splenectomy. Overall, among 35 patients (76.1%) who had splenectomy, 12

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Table I. Baseline characteristics of patients with giant splenic artery aneurysms (GSAAs) GSAA #10 cm (n ¼ 69) Variable

No.

Age, years, mean 6 SD Female sex

%

GSAA >10 cm (n ¼ 23) No.

54.4 6 17.4

%

Combined (N ¼ 92) No.

61.2 6 16.3

37

53.6

Proximal

24

34.8

Middle

22

31.9

10

% 56.1 6 17.3

43.5

47

51.1

4

17.4

28

30.4

13

56.5

35

38.0

Location

Distal or hilar

23

33.3

6

26.1

29

31.5

24

34.8

7

30.4

31

33.7

Essential hypertension

15

21.7

7

30.4

22

23.9

Portal hypertension

10

14.5

1

4.3

11

11.9

Pregnancy

10

14.5

2

8.7

12

13.0

Atherosclerosis

3

4.3

2

8.7

5

5.4

Inflammatory

1

1.4

2

8.7

3

3.3

Infection

2

2.9

0

0

2

2.2

Fibromuscular dysplasia

1

1.4

0

0

1

1.1

Iatrogenic

1

1.4

0

0

1

1.1

Syndromica

1

1.4

0

0

1

1.1

31

44.9

12

52.2

43

46.7

Ruptured aneurysm Presumed cause

Unknown or not reported b

Presentation

Abdominal pain

36

52.2

11

47.8

47

51.1

Shock

14

20.3

4

17.4

18

19.6

Gastrointestinal bleeding

11

15.9

3

13

14

15.2

Back or flank pain

4

5.8

3

13

7

7.6

Chest pain

1

1.4

0

0

1

1.1

13

18.8

7

30.4

20

21.7

Others

SD, Standard deviation. a Osteoarthritis-aneurysm syndrome. b Percentages do not add up to 100% because of overlapping as some patients had multiple causes or symptoms.

distal pancreatectomies, 5 gastric wedge resections, 2 segmental colectomies, and 1 gastrorrhaphy were performed concomitantly.

ENDOVASCULAR THERAPY Details of endovascular procedures are summarized in Table III. Overall, endovascular intervention was attempted in 39 patients and was successful in 35 (89.7%). Sandwich exclusion, which involves occlusion of the parent vessel proximal and distal to the aneurysmal neck, was the most commonly used endovascular procedure (19/ 39). This was performed with embolic agents only, such as coils, microcoils, vascular plug, and liquid agents; for aneurysms with anomalous origin, sandwich exclusion was mostly performed with covered stent placement in the superior mesenteric or common hepatic artery to occlude the inflow artery and embolization of the outflow artery (Supplementary Table, online only). Eleven patients had inflow occlusion, and one patient had outflow occlusion. Sac packing was employed as an

additional procedure in five patients and as a solo procedure in another four patients. Stent exclusion was successfully performed in four patients.

COMBINED THERAPY One patient had successful coil embolization followed by aneurysmectomy, splenectomy, and distal pancreatectomy for a middle-third GSAA. Another proximal aneurysm was successfully treated with coil embolization and surgical ligation with spleen preservation.

OUTCOMES ACCORDING TO THERAPEUTIC APPROACH Clinical features, aneurysm characteristics, and management outcomes were compared between the surgery group and the endovascular group (Table IV). The two groups did not differ in terms of sex, aneurysm size, multiple and ruptured aneurysms, management setting, success and reintervention rates, and aneurysm-related mortality. The mean follow-up for the surgery group and the endovascular group was 15.6 6 25.6 months and

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Fig. The numbers of cases treated with primary surgery (blue columns) and endovascular therapy (red columns) in 5-year blocks.

18.5 6 25.6 months, respectively. Patients in the surgery group were younger (P ¼ .056) and had lower overall postintervention complications (P ¼ .041). Three patients in the surgery group required further open surgical intervention for intra-abdominal bleeding or sepsis and drainage of pancreatic pseudocyst. In the endovascular group, four patients received either coil embolization or surgical salvage in the form of aneurysmectomy or splenectomy, with or without distal pancreatectomy, for incomplete endovascular hemostasis; one had splenectomy for splenic abscess; and another two had surgical débridement or multivisceral resection with liver transplantation for device-related infection. Overall, there was one periprocedural death in a middle-aged woman who died of a ruptured GSAA during surgery.

DISCUSSION True GSAAs are rare clinical entities, and their management poses a considerable challenge to both vascular surgeons and interventional radiologists. Our English literature search during a 15-year period yielded fewer than 100 cases,4-78 which supports the rarity of these lesions. To the best of our knowledge, this study is the first detailed analysis of clinical features and management of true GSAA >5 cm. Akbulut and Otan79 previously reported a review on GSAA $5 cm, but they included both true and false aneurysms without differentiation between the two entities in clinical presentation and management. Moreover, no attempt was made to analyze the different management options for true GSAA in terms of success rate and complications and to provide recommendations. The cause of GSAA varied with the size of the lesion. Aneurysms #10 cm were related to pregnancy and portal hypertension in 27.5% of cases. Other causes included essential hypertension, atherosclerosis, infection, postsplenectomy occurrence, fibromuscular dysplasia, and osteoarthritis-aneurysm syndrome.4,5,7,26,35,41 These

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results were in accordance with previously published series of common SAA.80,81 On the contrary, larger lesions (>10 cm) were related to pregnancy or portal hypertension in only 13% of cases. In fact, portal hypertension was more likely to be encountered as a consequence of GSAA due to mass effect or fistula formation (8.7%) rather than to be a cause (4.3%).9,11,43 This corroborates previous observations made by Long et al82 suggesting that many patients with GSAA >10 cm are typical of individuals in whom arterial aneurysms develop at sites other than the splenic artery. Our results suggest that GSAA has unique clinical features compared with smaller SAA. The mean age at presentation was 56 years, which is younger than the mean age of 61 years reported for common SAA.3,83 The difference probably reflects the larger proportion of symptomatic patients in our study population, who are often diagnosed at a younger age.84 Previous reports showed a female preponderance for common SAA3,81,83; however, the male to female ratio for GSAA was almost 1:1. This finding may relate in large part to the different underlying etiology and pathophysiology of GSAAs compared with smaller lesions. Common SAAs are typically asymptomatic and are incidental findings on abdominal imaging.3,81,83 Although abdominal pain is present in 78% to 100% of symptomatic SAA patients, in most cases, pain is not directly related to aneurysm.81,83 In stark contrast, almost three-fourths of GSAAs were symptomatic at the time of the initial diagnosis, and most, particularly distal lesions, had symptoms directly related to aneurysm. This includes symptoms due to mass effect on adjacent structures, secondary portal hypertension, and rupture. The rupture rate for GSAA was 34.8% compared with only 2% to 10% for common SAA.81,85 This finding supports the contention that the diameter of aneurysm correlates well with the risk of rupture. However, the overall mortality rate for ruptured GSAA was only 12.5%, which is much lower than the respective rates for smaller SAAs (20%-40%).3,80,86 This could partially be attributed to the different pattern of rupture. For smaller SAAs, rupture often occurs freely in the peritoneal cavity and is manifested with shock3,85; whereas in nearly half of ruptured GSAA cases, rupture was contained or the aneurysm eroded into adjacent veins or organs, leading to fistula formation and primarily manifested as stable gastrointestinal bleed.10-15 More important, most data in this study were derived from case reports and small case series, which usually report successful management and are subject to selection and publication bias. This review shows that GSAA has distinct anatomic characteristics. Whereas >90% of common SAAs are located in the distal two-thirds of the splenic artery,81,83 GSAAs were almost evenly distributed across the artery. One possible explanation for this distribution pattern is

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Table II. Details of surgical procedures in surgery group GSAA #10 cm (n ¼ 34) Surgical procedures

No.

GSAA >10 cm (n ¼ 13) %

No.

%

Open Ligation

2

5.9

3

Ligation with splenectomy

1

2.9

2

15.4

14

41.2

3

23.1

Aneurysmectomy with splenectomy

23.1

Aneurysmectomy, splenectomy, and gastrorrhaphy

1

2.9

0

0

Aneurysmectomy with multivisceral resectiona

7

20.6

5

28.4

Aneurysmectomy with end-to-end anastomosis

2

5.9

0

0

Aneurysmectomy and graft interposition

1

2.9

0

0

Aneurysmectomy and aortosplenic bypass

4

11.8

0

0

Laparoscopic Aneurysmectomy with splenectomy

1

2.9

0

0

Aneurysmectomy, splenectomy, and distal pancreatectomy

1

2.9

0

0

GSAA, Giant splenic artery aneurysm. a In addition to splenectomy, patients had distal pancreatectomy, gastric wedge resection, or segmental colectomy.

Table III. Details of endovascular procedures in endovascular group GSAA #10 cm (n ¼ 32) Procedures

No.

%

GSAA >10 cm (n ¼ 7) No.

%

Parent vessel flow preservation Sac packing only

3

9.4

1

14.3

Stent graft exclusion

4

12.5

0

0

Inflow occlusion only

6

18.8

4

57.1

Inflow occlusion and collateral embolization

0

0

1

14.3

No parent vessel flow preservation

Outflow occlusion and sac packing Sandwich exclusion only Sandwich exclusion and sac packing

1

3.1

0

0

14a

43.8

1

14.3

4

12.5

0

0

GSAA, Giant splenic artery aneurysm. a Including four patients with GSAAs with anomalous origin who had proximal stent placement in the superior mesenteric artery or common hepatic artery and coil embolization of the outflow artery.

that 21.4% of proximal GSAAs had anomalous origin, which is inversely associated with risk of rupture.87 On the other hand, middle and distal GSAAs had higher tendency to rupture, and therefore they may be underreported in the literature. Indeed, all lesions that were diagnosed during autopsy in this series were located in the middle or distal third of the splenic artery.21-23 Another interesting observation from this study is that aneurysms >10 cm had a predilection for the middle third of the splenic artery, which conforms to similar results in a previous study.4 The close anatomic relation of the pancreas with middle-third lesions may provide support to the wall and deter rupture of the aneurysm. Furthermore, the presence of an arteriovenous fistula in some cases allows aneurysms to drain in a relatively low pressure venous system, thus favoring growth of these lesions.45,49 Finally, in common with smaller SAAs,

GSAAs had mostly saccular morphology and were multiple in approximately 9% of the cases.3,81 Considering the rarity of GSAA, the diagnosis is often missed on initial clinical or endoscopic evaluation,19,20 and imaging studies are required to confirm diagnosis and to plan management. Common differential diagnoses include abdominal aortic aneurysms, gastrointestinal stromal tumors, pancreatic pseudocysts and tumors, and Dieulafoy lesion.12,16-20 Conventional ultrasound and Doppler ultrasound are inexpensive, noninvasive initial tests, but diagnostic accuracy is low. Contrastenhanced ultrasound is an alternative option with a higher sensitivity, particularly for ruptured aneurysms. Furthermore, it helps to characterize the aneurysm and to exclude presence of an arteriovenous fistula.62 Conventional CT and CT angiography were the most common minimally invasive imaging techniques employed

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Table IV. Comparison of surgery and endovascular groups Surgery group (n ¼ 47) Variable

No.

Age, years, mean 6 SD Female sex

Endovascular group (n ¼ 39) %

No.

53.2 6 16.4 26

% 60.2 6 16.8

55.3

17

P .056a

43.6

.279b .289b

Size #10 cm

34

72.3

32

82.1

>10 cm

13

27.7

7

17.9 .090c

Location Proximal

12

25.5

15

Middle

15

31.9

16

41.0

20

42.6

8

20.5

Multiple aneurysms

5

10.6

3

7.7

.724c

Ruptured aneurysm

14

29.8

14

36.8

.711b

Symptomatic presentation

36

76.6

25

64.1

.302b

Emergency setting

10

21.3

14

35.9

.132b

Success

46

97.9

35

89.7

.172c

6

12.8

12

34.2

.041b

Distal or hilar

38.5

Complications Overall Splenic infarction

e

8

Postembolization syndrome

e

2

Splenic abscess

e

1

Cardiac complications

e

1

Pulmonary complications

2

1

Device complications

1

2

Bleeding complications

1

1

Pancreatic complications

2

e

Acute renal failure

2

e

Other complications

4

e

Reintervention

3

6.4

7

15.8

.174c

Aneurysm-related mortality

1

2.1

0

0

1.000c

Follow-up, months, mean 6 SD

15.6 6 25.6

18.5 6 25.6

.595a

SD, Standard deviation. a Student t-test. b Pearson c2 test. c Fisher exact test.

for preoperative visualization of GSAA in our study, with diagnostic accuracy of 82% and 100%, respectively. Additional advantages of CT angiography include confirmation of the vascular trunk of origin in doubtful cases and more precise delineation of the aneurysm anatomy and its relationship with feeding vessel and the surrounding organs, particularly with use of threedimensional multiplanar reconstruction techniques. Contrast-enhanced magnetic resonance angiography was recently shown to be as sensitive as CT angiography in diagnosis as well as in follow-up of GSAA after surgical and endovascular intervention, particularly in the young age group.55 In addition, compared with CT angiography, it enables safer use of contrast material and imaging in any plane desired and does not involve ionizing

radiation.55,88 Of note, although considered the reference standard diagnostic method for SAA, invasive angiography was used to confirm diagnosis in only 40% of cases in this study. This is in line with recent observations that the advent of CT and magnetic resonance angiography has largely restricted the use of angiography to patients undergoing therapeutic endovascular intervention.88 Surgical intervention was the most commonly reported treatment modality for GSAA, with an overall success rate (98%) comparable to that of recent series.3,48 This approach is particularly beneficial in treating aneurysms associated with symptoms due to mass effect,6,8,10,11,44 hemodynamic instability, arteriovenous fistula, or concomitant other visceral aneurysms; in treating aneurysms >10 cm and infected aneurysms; and for distal lesions to

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avert potential risk of end-organ ischemia after endovascular embolization.89 The size and regional anatomy of GSAA, however, create obvious difficulties in surgical management. First, vascular control of the proximal splenic artery is at times difficult to obtain, which necessitates additional maneuvers, such as thoracoabdominal incision to secure proper exposure9 and medial rotation of the upper abdominal viscera to gain retroperitoneal access to the celiac trunk.4,82 Second, GSAAs were commonly associated with fistula formation or tight adherence to adjacent organs, which demanded additional visceral resection in 29% of patients undergoing surgery. The laparoscopic approach has recently been shown to be safe and feasible in treatment of SAA; it confers the advantages of lower morbidity, shorter hospital stay, and earlier recovery compared with open surgery.90 Moreover, a spleen preservation rate of up to 80% was reported with laparoscopic treatment.90 Nonetheless, it has rarely been used for management of GSAA. The two patients who were treated laparoscopically in our study had splenectomy because of distal location of the aneurysms.7,33 Previous studies have highlighted the importance of splenic preservation to prevent potential infectious complications. For proximal and middle-third aneurysms, aneurysmectomy with end-to-end anastomosis can be safely performed with preservation of the spleen. An alternative approach to aneurysmectomy is simple ligation, which was used in eight GSAA patients with a splenic preservation rate of 63%. In the presence of distal and hilar aneurysm location, surgical reconstruction of the artery can be impossible and splenectomy is unavoidable. In this study, the overall splenectomy rate for patients undergoing surgical treatment was 76%, regardless of the size of aneurysm, which was much higher than the rates of 17% to 43% reported for smaller aneurysms.3,48 Considering that more than half of these aneurysms were located in the proximal two-thirds of the splenic artery, the location would not simply explain the high rate of splenectomy. Other more important contributory factors were unsalvageable splenic injury during surgery, hypersplenism, presence of multiple aneurysms, and high rate of rupture. Treatment options for common SAAs have recently shifted from open surgery to the more cost-effective and less invasive endovascular therapy.91 Similarly, the endovascular approach has been employed in the management of GSAA during the last decade as a solo treatment or complementary to surgical intervention, with a technical success rate of 89.7%. This approach is particularly beneficial in elderly patients who are considered to be at high risk and for GSAA with anomalous origin. Compared with common SAA, endovascular treatment of GSAA is challenging because of the complex anatomy that often parallels these aneurysms. The size of these lesions may limit the efficacy of the endoluminal occlusion,

and vast quantities of embolic material, such as coils and liquid agents, may actually be required.67,73 Even more important in the endovascular treatment of these lesions is the presence of wide necks and tortuous splenic artery that renders isolation of the aneurysmal lumen from the circulation by a stent graft a difficult task. As a consequence, endovascular deconstructive techniques, which involve sacrifice of the splenic artery, were the most commonly employed. These include sandwich exclusion (aneurysm trapping) and inflow or outflow occlusion with or without sac packing. Of note, apart from the aneurysm size, none of the other aneurysm characteristics, such as location, presence of arteriovenous fistula, and multiplicity, were found to be determinant factors for the method of endovascular therapy. For lesions >10 cm, the majority were treated with inflow occlusion only. However, it is relevant at this juncture to mention that solely inflow occlusion is not recommended as it entails a high risk of technical failure due to retrograde reperfusion of the aneurysm through the collateral network of the short gastric arteries. On the other hand, seven of eight aneurysms for which a reconstructive method was used, either stent exclusion or isolated sac packing, were <10 cm. Isolated sac packing is reportedly associated with a high risk of coil compaction and recanalization in large aneurysms.92 Another potential disadvantage of the endovascular approach is the risk of splenic infarction, which may occur in as many as 40% of patients.64,83 Large splenic infarcts are more common with embolization of the distal third of the splenic artery. In this review, 21% of patients undergoing endovascular embolization had splenic infarction. Most of them recovered after conservative treatment, however, and only one required splenectomy because of the formation of splenic abscess.55 Even though endovascular intervention was inferior in terms of overall postintervention complications, it achieved comparable efficiency to surgery in reduction of aneurysm-related death and palliation of aneurysmrelated symptoms, particularly in cases in which surgery has been considered high risk. Notably, patients in the surgery group were younger and potentially healthier than patients in the endovascular group. Currently, with the growing use of CT and angiography, numerous aneurysms are being diagnosed in patients with poor clinical status. Such patients, who previously may have been excluded from surgery, would benefit from less invasive endovascular procedures. Most important, none of the patients who presented with ruptured GSAA and were treated with endovascular means died of aneurysmrelated causes. The authors speculate that the continually growing experience in combination with the development of novel devices, such as peripheral detachable coils, vascular plugs, and low-profile flexible stent grafts, will significantly improve the outcomes of endovascular treatment of GSAA in the years to come.

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Overall, GSAA is rare. However, the true incidence of GSAA may be underestimated. First, ruptured aneurysms diagnosed during autopsies might be missed as published studies generally favor reporting of patients presenting before rupture. Second, data collection was restricted to studies available in the English literature, and additional publications in the nonindexed and nonEnglish journals were not included. Third, we relied completely on published reports of whether aneurysms are true or false, and in case this was not clear, the report was excluded. Finally, cases of GSAA might have been included as part of other types of publications.93 However, the overall number of missed cases is likely to be small.

CONCLUSIONS GSAA is a rare clinical entity. These aneurysms are manifested at a younger age compared with common SAAs and have no sex predilection. Many GSAAs occur in individuals who are typical of patients affected by other nonsplenic aneurysms. The most commonly employed treatment of these vascular lesions remains aneurysmectomy and splenectomy, with or without pancreatectomy. A wide variety of endovascular therapeutic techniques are currently available for the treatment of GSAAs, demonstrating high technical success and low major complication rates, although the data regarding the long-term durability of these techniques could not be ascertained in our study. Moreover, the results of endovascular treatment in this report should be interpreted cautiously as failure of treatment would likely not be reported, and the success rate of endovascular management is probably overstated. A detailed and individualized analysis of each case in conjunction with sufficient understanding of the anatomy and hemodynamics of a particular aneurysm should guide the therapeutic decision. We would like to acknowledge Prof Gustavo Oderich, Prof Ahmed M. Elhassan, Prof Zhihui Dong, Prof Annette Thierauf-Emberger, Dr Federico Fontana, Dr Peter Stefanic, Dr Kemal Beksaç, Dr George Pitoulias, Dr Raymond Tang, Dr James Barnes, Dr Qi Liu, and Dr Vish Bhattacharya for sharing their experiences and cases in this study.

AUTHOR CONTRIBUTIONS Conception and design: HH, AS, GP, SS, RT, MT, RP Analysis and interpretation: HH, SS Data collection: HH, AS, GP, SS, RT, MT, RP Writing the article: HH, SS Critical revision of the article: HH, AS, GP, SS, RT, MT, RP Final approval of the article: HH, AS, GP, SS, RT, MT, RP Statistical analysis: Not applicable Obtained funding: Not applicable Overall responsibility: HH

2019

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Additional material for this article may be found online at www.jvascsurg.org.

Journal of Vascular Surgery -,

Number

I dentification

Volume

Hamid et al

10.e1

-

Records iden fied through database searching (n = 1704 )

Addi onal records iden fied through other sources (n = 5 )

Included

Eligibility

Screening

Records a er duplicates removed (n = 1458 )

Records screened (n = 1458 )

Records excluded (n = 1014 )

Full-text ar cles assessed for eligibility (n = 444 )

Eligible studies repor ng SAA > 5 cm (n = 81 )

Full-text ar cles excluded All true SAA < 5 cm 235 No size men oned 86 Duplicate cases 7 No dis nc on between true and false SAA 16 Others 19

Studies with inadequate informa on (n = 6 )

Studies included in the review (n =75 )

Total number of cases of GSAA > 5 (n = 92 )

Eligible unreported cases of GSAA > 5 cm (n = 2 )

Supplementary Fig (online only). Flow diagram. GSAA, Giant splenic artery aneurysm; SAA, splenic artery aneurysm.

Supplementary Table (online only). Endovascular materials used in patients who underwent sandwich exclusion Material

(N ¼ 19a)

Coils or microcoils

8

Coils or microcoils and liquid agent

5

Coils or microcoils and stent graft

3

Coils, liquid agent, and stent graft

1

Endovascular plug

1

Liquid agent only

1

This includes five splenic artery aneurysms (SAAs) with anomalous origin; four had stent grafting with embolization and one had coil embolization only.

2.

3.

4.

a

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