Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 2  |  Page : 179-183

Complex aortic aneurysm associated with multiple splanchnic arterial aneurysms and a giant coronary artery aneurysm: An endovascular nightmare


Department of Vascular Surgery, Institute of Vascular and Endovascular Sciences, Sir Ganga Ram Hospital, New Delhi, India

Date of Submission10-May-2020
Date of Decision09-Jun-2020
Date of Acceptance23-Jun-2020
Date of Web Publication13-Apr-2021

Correspondence Address:
Nikhil Vilas Chaudhari
Department of Vascular Surgery, Institute of Vascular and Endovascular Sciences, Sir Ganga Ram Hospital, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijves.ijves_55_20

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  Abstract 


A 50-year-old male diabetic, hypertensive and chronic smoker presented with complex aortic, splanchnic aneurysms with impending rupture of iliac artery aneurysm, underwent coiling of celiac artery, internal iliac artery, and fenestrated/chimney endovascular aortic repair. One month later, the patient landed in an emergency due to bleeding complications of common hepatic artery and gastroduodenal artery aneurysms, which were managed successfully using endovascular techniques. The mayhem of progressive nature of the disease in the patient continued as he had left lower limb ischemia due to occlusion of the iliac artery and iliac limb of the aortic graft device as he continued to smoke. All of the above sequences of events occurred over a period of 5 years. He also developed a giant aneurysm of left anterior descending of 6.8 cm diameter, which had to be repaired by open surgical technique. The patient has been followed up for 6 years since his first presentation, and he was managed successfully from all the catastrophe of events using endovascular techniques. Giant coronary artery aneurysm with previous aneurysms at multiple sites is quite unusual and rare. Currently, there is no consensus regarding the clinical characteristics, diagnostic method, and the treatment of these cases.

Keywords: Fenestrated/chimney endovascular aortic repair, giant coronary artery aneurysm, multiple aneurysms, pseudoaneurysm


How to cite this article:
Chaudhari NV, Bedi VS, Agarwal S, Yadav A, Satwik A, Srivastava A. Complex aortic aneurysm associated with multiple splanchnic arterial aneurysms and a giant coronary artery aneurysm: An endovascular nightmare. Indian J Vasc Endovasc Surg 2021;8:179-83

How to cite this URL:
Chaudhari NV, Bedi VS, Agarwal S, Yadav A, Satwik A, Srivastava A. Complex aortic aneurysm associated with multiple splanchnic arterial aneurysms and a giant coronary artery aneurysm: An endovascular nightmare. Indian J Vasc Endovasc Surg [serial online] 2021 [cited 2021 Jun 24];8:179-83. Available from: https://www.indjvascsurg.org/text.asp?2021/8/2/179/313557




  Case Report Top


In February 2014, a 50-year-old male patient, known case of diabetes mellitus and hypertension presented with pain abdomen on the right side for 8 days. Clinical examination revealed pulsatile mass palpable in the right lower quadrant of the abdomen, approximately 10 cm × 8 cm in size. The patient was a chronic smoker for 20 years, smoking 5–10 cigarettes per day. There was no history of fever, loss of appetite, and weight loss/asthenia. All peripheral pulses were palpable. Cutaneous stigmata of vasculitis, including digital ulceration and infarcts, were absent. There was no evidence of Marfanoid features or joint hypermobility. The cardiac examination was unremarkable. Neurological examination ruled out peripheral neuropathy. There was no evidence of any other swelling/bruit over the course of peripheral vessels.

Investigations

Ultrasound abdomen showed a large right iliac artery aneurysm with impending rupture. Computed tomography (CT) angiography was performed, which revealed a large aneurysm of the right common iliac artery (CIA) measuring 7.8 cm × 6 cm × 5.3 cm, involving internal iliac artery origin. There was a fusiform pararenal aortic aneurysm with a maximum diameter of 2.9 cm associated with aneurysms of the common hepatic artery (CHA) (3 cm × 2.4 cm) at its origin, and another smaller aneurysm (1.6 cm × 1.5 cm) was also seen at the origin of the right renal artery [Figure 1]. The inferior mesenteric and superior mesenteric artery (SMA) appeared to be anatomically normal. Laboratory investigations such as complete blood counts, renal function tests were normal. Erythrocyte sedimentation rate and C-reactive protein levels were marginally raised. Immunoglobulin G4 level was normal. Evaluation for underlying vasculitis, including antinuclear and antineutrophil cytoplasmic antibodies, was negative. Blood cultures performed in view of possible mycotic aneurysms were also persistently sterile. Echocardiography was normal with left ventricular ejection fraction of 55%. There was no evidence of any vegetations/nodules over heart valves.
Figure 1: Computed tomography angiography at initial presentation, (a) aneurysm of the origin of celiac artery (1.6 cm) and common hepatic artery (3 cm), (b) right iliac artery aneurysm (7.8 cm), (c) pararenal aortic aneurysm (2.9 cm), (d) right renal artery aneurysm (1.6 cm)

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Procedure details

Anatomy was unfavorable for the open repair as there were multiple associated aneurysms with patient's deteriorating clinical condition and impending risk of rupture of large iliac artery aneurysm. Complex endovascular aortic repair (EVAR) was performed as a life-saving surgery. The patient underwent physician modified (on table) fenestrated/chimney EVAR (left renal fenestration, right renal, and SMA-chimney). This achieved complete sealing with the exclusion of the aneurysm. Coil embolization of the celiac artery and the right internal iliac artery was done-using homemade Teflon coils. The procedure was performed in general anesthesia through bilateral femoral (open) and brachial (percutaneous) approach. The device used was Zenith Flex (Cook Medical, Bloomington, USA) aortic stent-graft (main body-28 mm × 96 mm, right limb-14 mm × 107 mm, left limb-14 mm × 90 mm), Stents used were Advanta (Getinge, Sweden) stents (Balloon expandable covered stent, 7 mm × 24 mm for the left renal, 8 mm × 59 mm for the right renal and 9 mm × 59 mm for the SMA) for the branches [Figure 2].
Figure 2: (a) Coiling of celiac artery, (b) superior mesenteric artery cannulation, (c) deployment of main device after positioning of branch vessel stent, (d) right iliac artery stent graft limb deployment, (e) angiogram postcomplex aortic repair, (f) exclusion of right iliac artery aneurysm

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Subsequent diagnoses and management of events

One month later, the patient developed sudden onset pain abdomen for lasting for 3–4 h and hypotension; on examination, tenderness and guarding were noted in epigastrium. Ultrasound abdomen suggestive of hemoperitoneum. Both lower limbs were warm, and distal pulses were palpable.

Computed tomography angiography

Previous aortic graft, right and left renal artery graft, and SMA graft were patent. There was no evidence of endoleak/branch vessel occlusion. Large ruptured pseudoaneurysm from the gastroduodenal artery (GDA) was identified with subhepatic and peripancreatic hematoma.

Procedure performed

As the patient was not hemodynamically stable, coiling and glue embolization of GDA pseudoaneurysm through stent grafted SMA cannulation through left brachial artery percutaneous approach under local anesthesia [Figure 3] As there was a persistent leak noted in the pseudoaneurysm sac on ultrasound evaluation the next day of the procedure, urgent bedside ultrasound-guided percutaneous injection of thrombin was done in the sac, till the cessation of the leak. The procedure was uneventful. The patient recovered from the shock and survived.
Figure 3: (a) Aneurysm of gastroduodenal artery, (b) cannulation of gastroduodenal artery aneurysm via superior mesenteric artery, (c) coil embolization of the aneurysm, (d) cyanoacrylate glue injection in the residual sac of the

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Subsequent events

After 1 year (September 2015), the patient developed progressive disabling claudication in the left leg and thigh; clinical examination revealed the absence of pulses in the left lower limb.

Procedure performed

Endovascular revascularization of the left lower limb-recanalization of the left limb of stent-graft device and left CIA stenting using a balloon-expandable bare metal stent proximally (Omnilink - 9 mm × 59 mm) and a self-expanding stent (Abbott-Absolute Pro - 10 mm × 100 mm), as shown in [Figure 4]. Postprocedure, distal pulses were palpable, and the patient was relieved of his symptoms.
Figure 4: (a) Left iliac artery occlusion, (b) balloon expandable stent deployment-Omnilink 9 mm × 59 mm, (c) extension using self-expanding stent 10 mm × 100 mm (d) final angiography

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Subsequent events

In June 2019, he presented with chest pain associated with palpitations and abdominal pain.

Investigations

Echocardiography s/o coronary artery aneurysm (CAA), CT angiography s/o large left anterior descending (LAD) aneurysm (6.8 cm × 6.2 cm) [Figure 5] and a CHA pseudoaneurysm. fluorodeoxyglucose positron emission tomography scan was performed on June 15, 2019, to look for uptake in other inflammatory sites/aneurysms in the body; however, no other pathology was detected.
Figure 5: (a) Large left anterior descending aneurysm with thrombus in the sac, (b) three dimensional reconstruction image, (c) thoracotomy and exposure of the aneurysm (d) left anterior descending aneurysm incision and repair in process

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Procedure performed

Thoracotomy with ligation of LAD and excision of the aneurysm and bypass using a saphenous vein graft was done by the cardiac surgery team. After stabilization of the patient, endovascular management of CHA aneurysm was performed successfully using coil embolization of the celiac artery just distal to previous coils, causing complete cessation of blood flow to the aneurysm through left brachial percutaneous approach under local anesthesia [Figure 6].
Figure 6: Coil embolization of celiac artery for common hepatic artery pseudoaneurysm

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Follow-up

The patient is asymptomatic at present till date April 20, 2020. He underwent contrast-enhanced ultrasound in August 2019, which was suggestive of patency of stent graft and all the branch vessels without evidence of endoleak. No de novo aneurysm detected so far.


  Discussion Top


The patient initially had an aortic aneurysm associate with multiple arterial aneurysms simultaneously involving the right iliac artery, right renal artery, and CHA. He presented to us with pain abdomen and a contained leak. With all the careful workup at his initial presentation and the possibility of the aneurysms being mycotic in nature, the decision for the appropriate intervention had to be made for the patient as there was implied risk of rupture of any of the aneurysms. Open surgical repair was not feasible given the probable mycotic etiology and anatomical difficulty in reconstructing all the vessels involved; hence, he was managed with the endovascular procedure at the time of his initial presentation. The subsequent disease progression landed him in the emergency room multiple times, and he was managed successfully using endovascular techniques, which gave him the advantages of being minimally invasive and less morbid compared to open surgical techniques for the same. Simultaneously, his progressive disease posed a significant technical challenge for endovascular surgeons.

On careful review of the available literature, there are case reports of multiple arterial aneurysms managed either with open or endovascular techniques.[1],[2] Multiple arterial aneurysms have been associated with a variety of disorders, including atherosclerosis,[3] vasculitides such as polyarteritis nodosa[4] and Behcet's disease,[5] infective mycotic aneurysms[6] and inherited conditions associated with a weakening of the arterial wall such as the Marfan and Ehler-Danlos syndromes.[7] In this case, the probable etiology was atherosclerosis, as other investigations were negative.

Regarding complications, all arterial aneurysms also carry a risk of spontaneous or traumatic rupture with catastrophic bleeding. This presentation is especially common with visceral aneurysms and is seen in more than half of all cases.[8] Maintaining a high index of suspicion is critical for ensuring timely therapy in the form of open repair or percutaneous intervention, both of which are approved strategies.[8]

In addition to pararenal aortic, iliac and multiple visceral arterial aneurysms, the patient developed a giant CAA, “metachronous” in nature. Review of literature suggests CAA is currently defined as a coronary artery dilatation >1.5 times the diameter of the normal adjacent segments or the diameter of the patient's largest coronary vessel,[9] and was reported to be 1%–4% of coronary angiography findings.[10] The reports of CAA combined with an additional aneurysm are quite common. However, CAA with concomitant aneurysms at multiple locations are quite unusual and rare,[11],[12] and is mostly limited to case reports and small case series. A systematic review of literature regarding CAA with coexisting aneurysms at multiple locations is available from Sichuan University, China.[2] Of the 76 patients, 53 (69.7%) of them had aneurysms in both coronary arteries, and 51 patients identified the concrete site of the CAA. O these, 32 occurred on the RCA (62.7%) and 22 occurred on the left main coronary artery (43.1%), 26 occurred on the LAD coronary artery (51%), 18 occurred on the left circumflex coronary artery (35.3%), and 35 (68.6%) had multiple CAAs. The main treatment of choice followed was surgical coronary artery bypass graft (CABG) and aneurysmal resection or ligation, percutaneous coronary intervention, or conservative drug treatment (anticoagulant and etiological treatment) In our case report, we noted giant CAA in LAD, which was repaired by aneurysmal resection and CABG.


  Conclusion Top


We present an interesting case here as no such case is reported in the past in the literature to the best of our review of the literature. Giant CAA was not concomitant with the aortic aneurysm here but, the aortic aneurysm was associated with the iliac artery, renal artery, and CHA aneurysms at his initial presentation. The patient subsequently presented with bleeding from an aneurysm of GDA, followed by the subsequent occurrence of giant CAA 5 years after the primary procedure. The highlight of the presentation here is the well-being of the patient and the success of the endovascular repair performed, evaluated over the period. We conclude; the anatomic morphology of the aneurysm and clinical characteristics should guide the specific treatment strategy of each individual case of multiple aneurysms.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Naha K, Vivek G, Shetty RK, Dias LS. Multiple idiopathic arterial aneurysms masquerading as aortic dissection. BMJ Case Rep 2013;2012-4.  Back to cited text no. 1
    
2.
Jiang LC, Cao JY, Chen M. Coronary artery aneurysm combined with other multiple aneurysms at multiple locations: A case report and systematic review. Medicine (Baltimore) 2017;96:e9230.  Back to cited text no. 2
    
3.
Göncü T, Sezen M, Vural H, Eris C, Yavuz S, Ozyazicioglu A. Sequential development of multiple atherosclerotic peripheral arterial aneurysms in a four-year period. Arch Med Sci. 2009;5:273-6.  Back to cited text no. 3
    
4.
Wang H, Li J, Jiang Y, Dai Q, Jiang Y, Hou Y, et al. Polyarteritis nodosa with multiple aneurysms and renal arteriovenous fistula successfully diagnosed by colour Doppler sonography. Clin Rheumatol 2013;32 Suppl 1:S89-92.  Back to cited text no. 4
    
5.
Calamia KT, Schirmer M, Melikoglu M. Major vessel involvement in Behçet's disease: an update. Curr Opin Rheumatol 2011;23:24-31.  Back to cited text no. 5
    
6.
Rojas A, Mertens R, Arbulo D, Garcia P, Labarca J. Multiple mycotic aneurysms due to penicillin nonsusceptible Streptococcus pneumoniae solved with endovascular repair. Ann Vasc Surg 2010;24:827.e5-8.  Back to cited text no. 6
    
7.
Mortani Barbosa EJ Jr., Pyeritz RE, Litt H, Desjardins B. Vascular Ehlers-Danlos syndrome presenting as rapidly progressive multiple arterial aneurysms and dissections. Am J Med Genet A 2011;155A: 3090-4.  Back to cited text no. 7
    
8.
Hirsch AT, Haskal ZJ, Hertzer NR, Bakal CW, Creager MA, Halperin JL, et al. ACC/AHA 2005 Guidelines for the management of patients with peripheral arterial disease (Lower Extremity, Renal, Mesenteric, and Abdominal Aortic): Executive Summary A Collaborative Report From the American Association for Vascular Surgery/Society for Vas. J Am Coll Cardiol 2006;47:1239-312.  Back to cited text no. 8
    
9.
Swaye PS, Fisher LD, Litwin P, Vignola PA, Judkins MP, Kemp HG, et al. Aneurysmal coronary artery disease. Circulation 1983;67:134-8.  Back to cited text no. 9
    
10.
Syed ML. Coronary artery aneurysm: A review. Prog Cardiovasc Dis. 1997;40:77-84.  Back to cited text no. 10
    
11.
Rigatelli G, Gemelli M, Franco G. Unusual combination of coronary artery, abdominal aortic and iliac artery inflammatory aneurysmal disease. Int J Cardiol. 2004;96:105–7.  Back to cited text no. 11
    
12.
Varol E, Ozaydin M, Altinbaş A, Akkuş I. Unusual combination of coronary artery, bilateral common carotid artery and left common iliac artery aneurysms. Anadolu Kardiyol Derg 2007;7:109-10.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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