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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 13  |  Issue : 1  |  Page : 27-30

Rare case of arterioportal fistula in a Nigerian; Presentation and successful management with transcatheter arterial embolization


1 IRDOC interventional Radiology Consulting Limited, Euracare Multispecialty Hospital, Victoria Island, Nigeria
2 ReMay Consultancy and Medical Services, Ikeja, Lagos State, Nigeria
3 Department of Radiology, Harbor-UCLA Medical Center, Los Angeles, California, USA
4 St. George's University School of Medicine, Grenada, Caribbean

Date of Submission24-Aug-2020
Date of Decision29-May-2021
Date of Acceptance08-Jun-2021
Date of Web Publication30-Jun-2021

Correspondence Address:
Dr. Aderemi Oluyemi
ReMay Consultancy and Medical Services, No. 24 Omotayo Ojo Street, Ikeja, Lagos State
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/njgh.njgh_24_20

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  Abstract 


Arterioportal fistula (APF) is a rare and potentially fatal cause of portal hypertension. In this case report, we present an unwonted case of a Nigerian with this disorder. As well, this article documents details of successful therapy for such a large APF with transcatheter arterial embolization which was carried out locally. This study is thus a first-of-its-kind for our African subregion. We hope that this article will help to highlight the role and significance interventional radiology services and expertise in an emerging health-care system as the one seen in West Africa – particularly as it relates to its benefits in the field of hepatology.

Keywords: Arterioportal fistula, embolization, interventional radiology, Nigeria, portal hypertension


How to cite this article:
Ninalowo H, Oluyemi A, Iwuchukwu CC, Olushekun AH. Rare case of arterioportal fistula in a Nigerian; Presentation and successful management with transcatheter arterial embolization. Niger J Gastroenterol Hepatol 2021;13:27-30

How to cite this URL:
Ninalowo H, Oluyemi A, Iwuchukwu CC, Olushekun AH. Rare case of arterioportal fistula in a Nigerian; Presentation and successful management with transcatheter arterial embolization. Niger J Gastroenterol Hepatol [serial online] 2021 [cited 2021 Oct 16];13:27-30. Available from: https://www.njghonweb.org/text.asp?2021/13/1/27/320305




  Introduction Top


Arterioportal fistulas (APFs) are abnormal communications between the hepatic artery and its branches and the portal vein. They are a rare cause of portal hypertension and can be fatal. A classification system has been proposed by Guzman et al., grouping APFs by size and etiology into Types 1 to 3.[1] Type 1 APFs are small, peripheral, intrahepatic fistulas with minimal physiologic consequences – these are most commonly caused by percutaneous liver biopsies while Type 2 AFPs are central with enough flow to result in elevated portal pressures. Rarely, APFs are congenital (Type 3). Early diagnosis and treatment with endovascular techniques can greatly improve patient outcomes. We present a case of a 26-year-old male with a large APF who was successfully treated with transcatheter arterial embolization (TAE) in Nigeria.


  Case Report Top


A 26-year-old male with a 5-month history of abdominal pain, distension, and jaundice was referred for interventional radiology (IR) management from the hepatologist. He had no prior history of alcohol ingestion, abdominal trauma, or liver biopsy. The patient had two sessions of endoscopic variceal banding for massive upper gastrointestinal bleeding from Grade 3 esophageal varices. Endoscopy had also shown portal hypertensive gastro- and duodenopathy. Hepatic Doppler showed an abnormally dilated left portal vein which showed mosaic appearance on Color Doppler interrogation and increased velocity (80.7 cm/s). There was direct communication between the left hepatic artery and left portal vein. Computed tomography (CT) imaging showed a peripheral 1.8 cm × 1.7 cm aneurysm from the left hepatic artery which connected directly to a large aneurysmal left portal vein with arterialization of the portal vein and its tributaries [Figure 1]. The celiac artery was diffusely enlarged with fusiform aneurysmal enlargement. There was arterialization of the portal vein tributaries with opacification of the mesenteric veins in the arterial phase. There was prominent flow within the inferior mesentery vein which continued down to the perirectal region. The liver span, outline, and biliary ducts were normal but there was heterogenous parenchymal echogenicity. The spleen was enlarged with homogenous echo pattern. Mild ascites were also noted. His hepatitides screens were all negative. Some relevant hepatology blood work results are listed in the composite [Table 1] which also includes those from 14-day and 2-month posttherapy for comparison.
Figure 1: (a) Preprocedure arterial phase computed tomography scan slide through the abdomen shows a markedly enlarged common hepatic artery (slim arrow) which is the same caliber as the aorta (arrowhead) at the same level. Also seen is early arterial opacification of some gastric varices (thick arrow), and ascites, suggesting portal hypertension. (b) A computed tomography cut shows a markedly enlarged left portal vein (between slim arrows) with early arterial opacification of the portal vein. Diffuse ascites (thick arrow)

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Table 1: Liver function test results

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Following review of his images, the decision was reached to proceed to the angiography suite the next day with a plan to occlude the left hepatic artery and essentially shut down the arterioportal shunt through TAE. The right common femoral artery was accessed using ultrasound guidance, over a 0.035 wire; a 6 Fr/45 cm braided sheath was advanced under active fluoroscopic monitoring. The 6Fr sheath was advanced into the proximal abdominal aorta and aortogram performed using 350 omnipaque contrast. Aortogram demonstrated significant hypertrophy of the celiac artery with hyperdynamic flow into the left hepatic artery and subsequently into the arterioportal shunt. The shunt led into a massive left portal vein aneurysm which subsequently fed arterialized flow into the portal system with hepatofugal flow. The left hepatic artery was subselected with a 5 Fr Bernstein-shaped angled catheter and arteriogram performed in multiple projections [Figure 2]. The 6 Fr sheath was advanced over the 5Fr catheter into the left hepatic artery and subsequently into the left hepatic artery aneurysm. A 12 mm Amplatzer plug was deployed within the aneurysm followed by multiple 8–10 mm fibered coils. Subsequently, a mixture of lipiodol and TruFill glue at a ratio of 1:1 was injected. Postembolization arteriogram showed complete cessation of flow within the shunt [Figure 3]. The entire procedure time was 56 min, and there were no immediate complications. He stayed in the hospital for 3 days with no recurrent gastrointestinal bleeding.
Figure 2: Acquired before embolization. (a) A catheter (thick arrow) is used to select the left hepatic artery and a microcatheter (slim arrow) is inserted into the proximal portion of the arterioportal fistula. When contrast in injected into the microcatheter, the length of the arterioportal fistula (curved arrow) is opacified. (b) The contrast then flows into a very large left portal vein (between arrowheads). From the large left portal vein, there is reversed flow in the large caliber main portal vein (slim arrow). Antegrade flow through the right portal vein (thick arrow) is not seen

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Figure 3: Posttherapy angiogram: showing successful embolization. After embolization, metal coil (thick arrow) can be seen lining and occluding the arterioportal fistula. Injection of contrast through a catheter in the left hepatic artery (thin arrow) shows that flow through the arterioportal fistula has been stopped. The larger left portal vein is not opacified. There is preferential flow into hepatic artery branches

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At the 2-week and 2-month follow-up, the patient had no evidence of gastrointestinal bleeding. He had increased independent mobility and resolution of bilateral lower extremity edema, abdominal swelling, and ascites. [Table 2] shows his laboratory results. CT abdomen and pelvis showed resolution of the APF [Figure 4].
Figure 4: Acquired a month postembolization: Axial computed tomography slices through the midabdomen demonstrate embolization coils in the fistula in the image on the left (slim arrow) with continued stasis and nonopacification of the enlarged left portal vein in the image on the right (thick arrow). The previously imaged gastric varies and ascites have resolved, suggest improved or resolved portal hypertension

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  Discussion Top


APFs can be caused by blunt or penetrating trauma to the liver (including iatrogenic causes), rupture of the hepatic artery, liver cirrhosis, infections, or neoplasms – less commonly, they can be of congenital origin or idiopathic.[1] In this patient, the APF could have been caused by rupture of the left hepatic artery aneurysm discovered on CT, or it may be congenital. The arterialization of many of the splanchnic veins, as well as the upper and lower gastrointestinal variceal bleeding, are indications of the extent of portal hypertension caused by the massive APF. Although TAE for severe APF is an effective treatment with minimal adverse effects, many patients with large APFs have required multiple embolization attempts, surgical treatment, or liver transplants to successfully eradicate the symptoms.[2],[3],[4],[5] Consequently, our prognosis of the index case was initially guarded given the severity of the APF. The patient was, however, successfully treated by a single TAE attempt with no postprocedure complications.

This case highlights the importance of the availability of IR in West Africa and other low-resource areas of the world.[6] Based on our literature research, there have been no previous reported cases of such a large APF treated in Nigeria and West Africa. This case presents a novel solution for such cases in the region. With endovascular techniques being less invasive and first-line techniques of choice for the treatment of APFs,[2] early access to IR services is important to avoid progression of the patient's condition such that more invasive procedures such as surgical correction or transplant surgery are required. This is especially important in Nigeria as these complex vascular surgical procedures are not readily available – and the needful IR services are only available in a handful of centers across the country.[6]


  Conclusion Top


This case report documents for the first time (to the best of our knowledge) the occurrence of a large APF in a Nigerian young man and details his successful management with TAE. The case highlights the importance of availability of IR in the field of hepatology and as an option for first-line treatment of APFs in West Africa, in a bid to reduce the need for surgery, and consequent morbidity and mortality from such APFs.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Guzman EA, McCahill LE, Rogers FB. Arterioportal fistulas: Introduction of a novel classification with therapeutic implications. J Gastrointest Surg 2006;10:543-50.  Back to cited text no. 1
    
2.
Kumar A, Ahuja CK, Vyas S, Kalra N, Khandelwal N, Chawla Y, et al. Hepatic arteriovenous fistulae: Role of interventional radiology. Dig Dis Sci 2012;57:2703-12.  Back to cited text no. 2
    
3.
Ward TJ, Marin ML, Lookstein RA. Embolization of a giant arterioportal fistula requiring multiple Amplatzer vascular plugs. J Vasc Surg 2015;62:1636-9.  Back to cited text no. 3
    
4.
Takata H, Makino H, Yokoyama T, Maruyama H, Hirakata A, Ueda J, et al. Successful surgical treatment for intrahepatic arterioportal fistula with severe portal hypertension: A case report. Surg Case Rep 2019;5:67.  Back to cited text no. 4
    
5.
Takagi K, Yagi T, Yoshida R, Shinoura S, Umeda Y, Nobuoka D, et al. A successful case of deceased donor liver transplantation for a patient with intrahepatic arterioportal fistula. Hepatol Res 2016;46:1409-15.  Back to cited text no. 5
    
6.
Ninalowo, H. We are Simply on a Mission: Interventional Radiology is Key to Moving Medicine Forward in Nigeria. Available from: https://interventionalnews.com/nigeria-interventional-radiology/. [Last accessed on 2020 Aug 12].  Back to cited text no. 6
    


    Figures

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