Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 9  |  Issue : 5  |  Page : 407-410

Successful management of abdominal aortocutaneous fistula following emergency endovascular aneurysm repair for aortoenteric fistula in a patient with Takayasu arteritis


1 Department of Vascular and Endo-Vascular Surgery, Kauvery Hospital, Chennai, Tamil Nadu, India
2 Department of Cardiology, Kauvery Hospital, Chennai, Tamil Nadu, India

Date of Submission17-Jul-2022
Date of Decision02-Aug-2022
Date of Acceptance04-Aug-2022
Date of Web Publication13-Jan-2023

Correspondence Address:
Dr. Rahul Ralph Sima
Department of Vascular and Endo-Vascular Surgery, Kauvery Hospital, Chennai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijves.ijves_45_22

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  Abstract 


Aortocutaneous fistula is an extremely rare complication after aortic endograft infection. We report on the management of a patient who presented with aortocutaneous fistula following emergency endovascular aneurysm repair (EVAR) of an aortoenteric fistula (AEF). A 50-year-old female with Takayasu arteritis who had undergone thoracic endovascular aortic repair (TEVAR) for a thoracic aortic aneurysm, 23 years ago presented with a spontaneous abdominal AEF and another contained ruptured descending thoracic aortic aneurysm distal to the previous endograft. She underwent emergency bridge EVAR and redo-TEVAR. Thirteen months following EVAR, she presented with endograft infection and a spontaneous aortocutaneous fistula. She was successfully managed with an axillo-unifemoral bypass followed by EVAR endograft explantation. She was discharged on long-term antibiotics and has been symptom free for 12 months.

Keywords: Aortocutaneous fistula, aortoenteric fistula, endograft infection, Takayasu arteritis


How to cite this article:
Sima RR, Sekar N, Gopalamurugan A B, Rajan A, Sebastian JJ. Successful management of abdominal aortocutaneous fistula following emergency endovascular aneurysm repair for aortoenteric fistula in a patient with Takayasu arteritis. Indian J Vasc Endovasc Surg 2022;9:407-10

How to cite this URL:
Sima RR, Sekar N, Gopalamurugan A B, Rajan A, Sebastian JJ. Successful management of abdominal aortocutaneous fistula following emergency endovascular aneurysm repair for aortoenteric fistula in a patient with Takayasu arteritis. Indian J Vasc Endovasc Surg [serial online] 2022 [cited 2023 Jan 29];9:407-10. Available from: https://www.indjvascsurg.org/text.asp?2022/9/5/407/367719




  Introduction Top


Aortoenteric fistula (AEF) after aortic intervention is rare and carries a high morbidity and mortality. Bridge endovascular therapy is being increasingly used for damage control after AEF. Greater use of emergency aortic endovascular interventions has led to increased incidences of endograft infection. Aortocutaneous fistula is a dangerous, extremely rare complication which has only been reported following sternal wound infection after open thoracic aortic surgery. This complication has been reported previously only once after abdominal aortic endograft infection. This rare and dangerous complication presents a challenge to manage. We present a case of an aortocutaneous fistula following bridge endovascular aneurysm repair (EVAR) done for an AEF which was managed successfully.


  Case Report Top


A 50-year-old female was referred to our hospital with complaints of abdominal and back pain and massive lower gastrointestinal bleeding.

She had a long past medical history starting from when she had been diagnosed with Takayasu arteritis (TA) at the age of 26 years during her first pregnancy. At the age of 27 years, she was subsequently diagnosed to have a saccular aneurysm of the descending thoracic aorta, which was managed by thoracic endovascular aortic repair (TEVAR) at another hospital. Since her femoral and iliac arteries were small in caliber, an infra-renal aortic conduit using a 10-mm polytetrafluoroethylene graft (Gore-tex graft, W.L. Gore Inc) was created for TEVAR deployment, which was closed later as a patch. Postprocedure, complete sac thrombosis was noted, and she was maintained on antihypertensives and immunosuppressants. Her inflammatory markers were normal at the time. She was noted to have concomitant left subclavian and bilateral renal artery stenosis during the procedure. She was followed up for 5 years after which she was lost to follow-up. She presented 23 years after TEVAR with massive lower gastro intestinal bleeding and was referred to our hospital.

She was disoriented with a blood pressure of 90/60 mmHg and her hemoglobin was 2.8 g/dL. She was resuscitated, and a computed tomography (CT) scan carried out, showed a saccular pseudoaneurysm of the distal abdominal aorta proximal to the bifurcation which was communicating with the distal ileum in the right iliac fossa. There was a contained rupture of another aneurysm in the descending thoracic aorta at the distal end of the previously implanted thoracic endograft.

In view of her poor general condition and aneurysms at two sites, she was considered high risk for open surgical repair and was taken up for emergency endovascular repair. Through an open right femoral access, a stent graft 34 mm × 32 mm × 167 mm (Valiant, Medtronic) was deployed across the distal thoracic aortic aneurysm landing above the celiac artery. A bifurcated EVAR device (Endurant, Medtronic) was deployed to cover the abdominal aortic pseudoaneurysm and AEF. She recovered well and was advised laparotomy and bowel repair. However, since the patient symptomatically improved, she refused surgery and was managed conservatively on long-term antibiotics without definitive bowel repair. At two months, a follow-up CT showed no endoleak or enteric fistula. Four months later, she developed a spontaneous wound in the right groin (right femoral artery access site), which discharged pus for which periodic dressing and antibiotic therapy was administered at a local hospital.

She was on follow-up at a local hospital but after a year presented with abdominal pain, fever, pus discharge, and bleeding from the right groin wound. Her left lower limb pulses were normal, but right femoral and distal pulses were absent with right ankle–brachial index (ABI) of 0.6. Wound and blood cultures grew no organism. CT aortogram showed an infected infrarenal aortic stent graft with a fistulous tract extending from the endograft to the right groin [Figure 1]a and [Figure 1]b. However, there was no enteric communication. In view of the infected endograft and worsening sepsis, she was planned for a left axillofemoral bypass and explantation of the EVAR graft. A left axillo-unifemoral bypass using 7-mm ringed polytetrafluroethylene graft (Gore-tex, W.L. Gore Inc) was carried out followed by midline laparotomy. On laparotomy, a thick-walled abscess cavity was seen adherent to the aorta with its anterolateral wall sloughed off exposing the stent graft in the cavity [Figure 2]. Cecum and terminal ileum were adherent to the cavity, but there was no fistulous communication. A fistulous tract lined by chronic granulation tissue was seen extending from the aortic endograft to the right groin. The right limb of the endograft was seen filled with infected thrombus and surrounded by chronic granulation. Proximal end of the endograft was dislodged and the entire graft removed in toto [Figure 3]. Aortic stump was suture ligated, taking care to preserve the renal arteries. The abscess cavity was curetted and packed with a pedicled omental flap. Both common iliac arteries were ligated proximal to the internal iliac arteries to preserve cross pelvic collateral flow to the right lower limb.
Figure 1: (a) CT showing gas inside and outside the right limb of the stent graft and a collection anterolateral (b) Thrombosed right limb of the endograft with an abscess tracking along the iliac vessels up to the groin (arrow). EVAR: Endovascular aneurysm repair, CT: Computed tomography

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Figure 2: Abscess cavity with sloughed off anterolateral wall of aorta, exposing the endograft and containing thick infected granulation

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Figure 3: Explanted endograft. Part of theadherent intima attached to the proximal bare stent and barbs is also seen

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The patient had an uneventful postoperative recovery. She had palpable pulses in her left leg and her right leg ABI was 0.6. Cultures from intraoperative tissue did not grow any organism. She was discharged on dual antiplatelets and long-term broad-spectrum antibiotics. The cutaneous fistulous opening healed in 2 months. At 1-year follow-up, she was ambulant and symptom free.


  Discussion Top


The prevalence of TA in Indian patients has been reported in numerous Indian studies with a female-to-male ratio of 1.58:1.[1] The incidence of aortic aneurysm in TA has been reported to be around 10%–45% and commonly affects the descending thoracic and thoracoabdominal aorta.[2],[3] Aneurysms in TA are usually found in patients above 40 years of age and are rarely reported at a younger age as was seen in our patient.

Endovascular repair is now routinely offered for degenerative thoracic aneurysms since the procedure is less invasive and has a lower complication rate. Endovascular repair of TA aneurysms has been rarely reported. Treatment of aneurysms in TA requires a therapeutic strategy different from that of degenerative aneurysms due to the diffuse, progressive, and relapsing nature of the disease. Our patient is probably one of the first patients to undergo a TEVAR for a descending thoracic aortic aneurysm in India.

The formation of recurrent aneurysms in the landing zones after TEVAR is not commonly seen in degenerative aneurysms, but has been reported after TEVAR for TA.[4] The presence of the distal aneurysms may be due to the prolonged, relapsing, inflammatory nature of TA. The causes for AEF after aortic intervention are mechanical contact of graft material and bowel, leading to erosion of the intestinal wall, retroperitoneal contamination, or an inflammatory process as seen in aortoarteritis.[5] Our patient had a prosthetic patch at the distal aorta just above the bifurcation, which possibly was the site of a recurrent aneurysm, leading to an AEF 23 years later. The meta-analysis by Kakkos et al. reported that the common symptoms of AEF are bleeding (71.7%) and hemorrhagic shock (33.1%) as were seen in this patient.[6] The most common location of the fistula is at the duodenum and rarely large bowel is involved.[6] Emergency open surgery for AEF has rather dismal outcomes and endovascular bridge techniques are being preferred frequently in these critical patients.

Since this patient presented with hemorrhagic shock and multiple aneurysms with contained rupture, endovascular bridge approach was carried out for AEF. This patient was referred back to the treating surgeon for laparotomy and evacuation of hematoma with repair of the bowel, but the patient refused further surgery since she was asymptomatic. She was, however, maintained on long-term antibiotics. A meta-analysis of endovascular management of AEF showed that AEFs were managed successfully in 93% with a mortality of 8.5% at 30 days. However, follow-up of these patients showed recurrence and sepsis in 52% of patients at 12 months, if they did not have subsequent bowel repair surgery.[6] The overall survival was reported as 68% at the end of 12 months.[6] Evidence of sepsis preoperatively was indicative of poor outcome. Secondary AEFs as compared to primary, have a threefold increased risk of persistent/recurrent infection.[7],[8] Abdominal aortic stent graft infection is estimated to range from 0.5% to 0.7%.[9]

Aortocutaneous fistula is a rare complication following sternal infection in open thoracic aortic surgery. This dreaded complication has been reported once previously following infra-renal abdominal EVAR endograft infection. In this case report, the authors conclude that it was advisable to explant the infected endograft to improve patient outcomes in similar situations.[9] The recommended treatment for aortic endograft infection is removal of the infected graft and revascularization. Aortic reconstruction can be in situ using deep vein grafts, homografts or antibiotic-impregnated prosthetic grafts, or extra-anatomical axillofemoral bypass with prosthetic graft.

Mortality rate is extremely high after surgical treatment in these patients with an infected endograft regardless of the reconstructive method, material used, or clinical condition.[10] In our patient, an extra-anatomical bypass with removal of the infected EVAR stent graft and ligation was well tolerated. This is probably the first case report on successful management of an aortocutaneous fistula.


  Conclusion Top


Aortocutaneous fistula is an extremely rare complication of abdominal aortic endograft infection. This complication can be managed successfully by staged extra-anatomical bypass and endograft explantation. This complication could have been avoided if bowel repair had been undertaken after EVAR.

Declaration of patient consent

The authors certify that they have obtained an appropriate patient consent form. In the form, the patient has given her consent for her images and other clinical information to be reported in this journal. The patient understands that her name and initials will not be published, and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Chhetri MK, Raychaudhuri B, Neelakantan C, Basu J, Chaki S, Saha AK. A profile of non-specific arteritis as observed in Eastern India. J Assoc Physicians India 1974;22:839-47.  Back to cited text no. 1
    
2.
Robbs JV, Human RR, Rajaruthnam P. Operative treatment of nonspecific aortoarteritis (Takayasu's arteritis). J Vasc Surg 1986;3:605-16.  Back to cited text no. 2
    
3.
Kieffer E, Chiche L, Bertal A, Koskas F, Bahnini A, Blã Try O, et al. Descending thoracic and thoracoabdominal aortic aneurysm in patients with Takayasu's disease. Ann Vasc Surg 2004;18:505-13.  Back to cited text no. 3
    
4.
Regina G, Bortone A, Impedovo G, De Cillis E, Angiletta D, Marotta V. Endovascular repair of thoracic stent-graft bulging rupture in a patient with multiple Thoracic Aneurysms due to Takayasu disease. J Vasc Surg 2007;45:391-4.  Back to cited text no. 4
    
5.
Miyata T, Sato O, Deguchi J, Kimura H, Namba T, Kondo K, et al. Anastomotic aneurysms after surgical treatment of Takayasu's arteritis: A 40-year experience. J Vasc Surg 1998;27:438-45.  Back to cited text no. 5
    
6.
Kakkos SK, Papadoulas S, Tsolakis IA. Endovascular management of arterioenteric fistulas: A systemic review and meta-analysis of the literature. J Endovasc Ther 2011;18:66-77.  Back to cited text no. 6
    
7.
Antoniou GA, Koutsias S, Antoniou SA, Georgiakakis A, Lazarides MK, Giannoukas AD. Outcome after endovascular stent graft repair of aortoenteric fistula: A systematic review. J Vasc Surg 2009;49:782-9.  Back to cited text no. 7
    
8.
Arima D, Suematsu Y, Kurahashi K, Shimizu T, Nishi S, Yoshimoto A. Recurrence of aortoenteric fistula after endovascular aortic repair. Ann Vasc Dis 2020;13:90-2.  Back to cited text no. 8
    
9.
Ziaja D, Biolik G, Chudek J, Ziaja K. Purulent cutaneous fistula: As the first symptom of the late aortic stent-graft infection-A case report and review of the literature. Case Rep Surg 2013;2013:421780.  Back to cited text no. 9
    
10.
Sharif MA, Lee B, Lau LL, Ellis PK, Collins AJ, Blair PH, et al. Prosthetic stent graft infection after endovascular abdominal aortic aneurysm repair. J Vasc Surg 2007;46:442-8.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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