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Portal Vein Thrombosis after Endovascular Embolization of Splenic Artery for a Symptomatic Arterioportal Fistula: A Case Report and Literature Review
Accepted Manuscript Portal Vein Thrombosis after Endovascular Embolization of Splenic Artery for a Symptomatic Arterioportal Fistula: a Case report and Literature review Pyeong Hwa Kim, Dong Il Gwon, Heung Kyu Ko PII:
S0890-5096(18)30585-5
DOI:
10.1016/j.avsg.2018.05.075
Reference:
AVSG 3984
To appear in:
Annals of Vascular Surgery
Received Date: 18 April 2018 Accepted Date: 1 May 2018
Please cite this article as: Kim PH, Gwon DI, Ko HK, Portal Vein Thrombosis after Endovascular Embolization of Splenic Artery for a Symptomatic Arterioportal Fistula: a Case report and Literature review, Annals of Vascular Surgery (2018), doi: 10.1016/j.avsg.2018.05.075. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Full Title Page
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Manuscript title: Portal Vein Thrombosis after Endovascular Embolization of Splenic
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Artery for a Symptomatic Arterioportal Fistula: a Case report and Literature review
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Manuscript type: Case report
Authors:
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Pyeong Hwa Kim
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Dong Il Gwon
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Heung Kyu Ko
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Department of Radiology, Asan Medical Center, College of Medicine, 88 Olympic-ro
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43-gil, Songpa-gu, Seoul 05505, Korea
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†
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Department of Radiology, Asan Medical Center, Ulsan University College of Medicine,
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88 Olympic-ro 43-gil, Songpa-gu, Seoul 05505, Korea
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Telephone: 82-2-3010-3974; Fax: 82-2-3010-6645; E-mail: [email protected]
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Corresponding author: Heung Kyu Ko
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E-mail list:
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Pyeong Hwa Kim: [email protected]
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Dong Il Gwon: [email protected]
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Heung Kyu Ko: [email protected]
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Acknowledgement: None.
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Disclosure statement:
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Funding: None.
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Conflict of Interest: The authors declare that they have no conflict of interest.
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Ethical approval: For this type of study formal consent is not required.
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Informed consent: The requirement to obtain written informed consent was waived from
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the IRB of Asan medical center.
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Portal Vein Thrombosis After Endovascular Embolization of the Splenic Artery for
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a Symptomatic Arterioportal Fistula: Case Report and Literature Review
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Abstract Arterioportal fistula (APF) can induce severe portal hypertension, and therefore
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requires appropriate and timely management. Endovascular treatment is increasingly
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used for the treatment of APFs due to its minimally invasive nature, although this
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procedure can lead to potentially fatal portal vein thrombosis (PVT). Reports of this
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complication are, however, rare. Here, we describe the case of a 65-year-old woman
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who experienced an extensive thrombosis from the splenic vein to the right portal vein
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following embolization of a splenic APF, leading to the requirement for ICU care and a
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prolonged hospital stay. In addition, the literature was reviewed to describe the clinical
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manifestations, diagnosis, and treatment of PVT after embolization of APF.
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Introduction Arterioportal fistulas (APFs), characterized by an abnormal communication
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between the systemic arteries and the portal vein, are rare but can be fatal. A systematic
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review of 88 patients with APFs reported the most frequent cause of APF to be trauma
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(28%), followed by iatrogenic causes (16%), congenital causes (15%), tumor (15%),
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and rupture of aneurysms (14%). Most APFs originated from the hepatic artery (65%),
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followed by the splenic (11%) and superior mesenteric arteries (10%).1 Because APFs
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can induce severe portal hypertension with multiple morbidities, appropriate and timely
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management is essential.
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Endovascular treatment is increasingly used for the treatment of intrahepatic
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and extrahepatic APFs because of low cost, repeatability, and minimally invasive
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nature.1-3 While some reports describe cases of extensive portal vein thrombosis (PVT)
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after embolization of mesenteric, splenic, or intrahepatic arteriovenous fistulas,4-6 this
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rare, but serious, complication remains poorly described.
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This report documents a case of PVT after the embolization of an APF and
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reviews the literature to identify common clinical manifestations of this condition, as
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well as its diagnosis and treatment.
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Case report This study was approved by the institutional review board (IRB); the
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requirement for informed consent was waived because of the retrospective nature of the
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case report. From January 1990 to October 2017, nine patients underwent embolization
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to treat an APF. Of those, one patient subsequently experienced a symptomatic PVT.
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A 65-year-old woman was referred to our institution due to progressive
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abdominal distension, dyspnea, and intermittent hematochezia during the previous
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month. At admission, a percutaneous peritoneal drainage catheter was inserted to drain
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ascites. The patient was alert, with a blood pressure of 144/92 mmHg and tachycardia
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(103 bpm). There was no history of heavy alcohol use. The only notable laboratory
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finding was an elevated serum creatinine level (3.74 mg/dL), necessitating dialysis.
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Other laboratory tests (including platelet count, prothrombin time, serum aspartate [AST]
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and alanine aminotransferases [ALT] concentration, bilirubin, alkaline phosphatase, and
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γ-glutamyl-transferase) were within the normal range. The patient was seronegative for
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hepatitis C viruses and positive for antibodies against hepatitis-B surface antigen.
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Contrast-enhanced computed tomography (CT) scan of the abdomen and pelvis
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showed the following: i) markedly dilated splenic vein with early opacification of the
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splenic and portal veins; ii) slightly dilated splenic artery; iii) homogeneously decreased
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attenuation of the spleen suggestive of splenic infarction; iv) a large amount of ascites;
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and v) mesenteric venous congestion suggestive of ischemic colitis (Figure 1). Surface
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nodularity of the liver, indicating liver cirrhosis, was not observed. Volume rendering
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images showed a direct communication between the splenic artery and vein, leading to a
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profuse inflow of blood to the liver and causing severe portal hypertension. A large
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volume of ascites was considered to be secondary to the portal hypertension. Acute
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history of medical renal disease and the patient’s kidneys and urinary tract were seen to
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be normal on CT scanning. Intermittent hematochezia was considered to be due to
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ischemic colitis, which was confirmed by colonoscopic biopsy.
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To isolate the splenic artery and portal venous system, angiographic
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management of the fistula was considered appropriate due to its minimally invasive
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nature. Selective splenic arteriography via the right femoral artery using a 5F Cobra II
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catheter (Cook Medical, Bloomington, IN) showed a fistula between the splenic artery
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and the markedly dilated splenic vein. The portal vein was not opacified in the venous
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phase of subsequent superior mesenteric arteriography. However, both direct
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embolization of the arteriovenous fistula and stent graft failed due to the strikingly high
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flow velocity, tortuosity of the splenic artery, and complex vascular anatomy. Because
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the spleen was totally infarcted, proximal splenic arterial embolization was therefore
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performed, using a 2.4F microcatheter (Boston Scientific Corp., Natick, MA) with
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multiple Interlock-18 detachable coils (Boston Scientific Corp.), one measuring 6 mm ×
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20 cm, two measuring 10 mm × 30 cm, and two measuring 20 mm × 50 cm. Histoacryl
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(B. Braun, Melsungen, Germany) was used in a 1:2 mixture with lipiodol. After
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completion of embolization, the fistula and splenic vein were not opacified on celiac
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arteriography and patent portal flow was observed during the venous phase of superior
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mesenteric arteriography, indicating successful occlusion of the splenic APF (Figure 2).
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After completion of the embolization procedure, the patient was closely
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observed in the intensive care unit. While the patient’s abdominal distension was
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initially relieved, distension recurred 1 week later. Follow-up CT 9 days after the
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embolization showed diffuse thrombosis of the splenic vein extending to the right
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ALT (24 to 56 U/L), alkaline phosphatase (98 to 245 IU/L), and total bilirubin (0.9 to
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4.4 mg/dL). At that time, creatinine was within normal range. The patient complained of
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pain in both legs, and duplex sonography showed extensive deep vein thrombosis (DVT)
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in the right femoral vein and the left posterior tibial vein. At that time, thrombocytosis
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(404,000/mL) was also noted. Since a hematologic disorder was suspected, bone
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marrow biopsy was performed, but the only remarkable finding was an increased
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number of megakaryocytes. No mutations were detected in JAK-2, MPL, and CALR
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gene studies. Therefore, the high platelet count was considered to be due to reactive
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thrombocytosis. To prevent the progression of PVT and DVT, heparin therapy was
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initiated (20,000 IU/day), and an IVC filter was inserted to prevent pulmonary
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thromboembolism. Follow-up CT 1 month after the embolization (2 weeks after
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initiating heparin therapy) showed a decreased PVT volume. However, discharge was
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delayed for a month due to the following comorbidities: i) recurrent episodes of fever
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caused by spontaneous bacterial peritonitis requiring antibiotic therapy and external
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drainage; ii) severe leg swelling and pain caused by DVT; and iii) post-AKI diuresis
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requiring hydration. Two months after embolization, CT scanning showed a further
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decrease in thrombus volume and no residual ascites (Figure 4). The patient was
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subsequently discharged with warfarin treatment. At the last follow-up (5 months after
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embolization), the patient remained clinically stable with no symptoms of portal
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hypertension. There were no recurrent episodes of hematochezia and abdominal
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distension. Creatinine levels were within normal range, and the extent of the DVT was
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further decreased. The patient’s platelet count remained elevated (510,000/mL), but this
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was considered likely to be reactive considering the unremarkable bone marrow biopsy
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results and infarcted spleen.
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Discussion APF is defined as an anomalous communication between the splanchnic artery
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and the portal venous system. It can be classified by location (intrahepatic or
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extrahepatic), or etiology (congenital or acquired). The most common cause of acquired
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APF is trauma (28%), primarily after a penetrating injury.7 Other causes include
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iatrogenic causes (16%), rupture of splanchnic aneurysms (14%), and, less frequently,
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cirrhosis and hepatic malignancy.1 In general, small and peripherally located APFs are
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asymptomatic and do not require therapeutic intervention. By contrast, large, centrally
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located APFs with high shunt flow are often symptomatic, inducing portal hypertension
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and requiring treatment.8 Despite the lack of data from large prospective studies,
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endovascular embolization is widely used for the treatment of symptomatic APFs
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because of its technical feasibility and good safety profile.6,9-11
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The patient described in this report experienced an extensive PVT following
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embolization of an APF with several related comorbidities. In this case, proximal
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splenic arterial embolization, rather than stent graft or direct embolization of the fistula,
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was performed. Although stent graft has the advantage of preserving arterial access, it is
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technically challenging due to the tortuous nature of the splenic artery, high flow
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velocity of the fistula, and absence of a distal landing zone.10,12 After the proximal
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splenic artery embolization, splenic venous flow became sluggish, inducing an
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extensive PVT. Right portal vein obliteration and multiple associated comorbidities
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necessitated a prolonged hospital stay (2 months). Therefore, greater awareness of this
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potentially fatal complication is required for better management and prevention.
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ACCEPTED MANUSCRIPT A meticulous literature search identified reports of 11 patients who had
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experienced PVT after embolization of APF (Table 1).4-6,13-17 Eight (66.7%) of these
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APFs were iatrogenic. The median interval from embolization to PVT diagnosis was 7.5
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days (range, 2 days to 3 years; interquartile range, 3.3–13 days). Reported clinical
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manifestations of PVT after embolization included abdominal pain, diarrhea, abrupt
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increases in bilirubin and creatinine levels, gastrointestinal bleeding, and hepatic
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encephalopathy. In 7 of the 12 patients, PVT was detected on follow-up imaging 2–10
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days after embolization, without clinical manifestation.
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In seven (87.5%) of eight patients, anticoagulation therapy alone was effective
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for treating PVTs. Anticoagulation therapy is a well-known, basic treatment for
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symptomatic PVT, recommended both by the American College of Chest Physicians
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(ACCP) and the American Association for the Study of Liver Disease (AASLD).18,19
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According to one systematic review,20 52.3% (116/228) of patients with PVT treated
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with anticoagulation had a complete (37.6%; 85/228) or partial (13.6%; 31/228)
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recanalization, with a low short-term complication rate of 1.3% (3/228, comprising
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minor retroperitoneal bleeding [n = 1], epistaxis [n = 1], and gum bleeding [n = 1]).
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Therefore, anticoagulation therapy can be an initial option with minimal safety concerns.
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Of note, a delay in initiating anticoagulation therapy is an independent predictor of
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recanalization failure.21 Therefore, prompt CT for the patients with recurred ascites,
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abdominal pain, or increased bilirubin and creatinine levels may be required for early
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detection of PVTs, thereby increasing the likelihood of successful recanalization.22,23
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Two patients (Table 1, patients 4 and 5) with extensive PVTs involving both the
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intrahepatic and extrahepatic portal vein required additional treatment, such as
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transjugular intrahepatic portosystemic shunt (TIPS) or thrombolysis. Thrombolysis is
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experienced a complete (40.8%; 29/71) or partial (45.1%; 32/71) recanalization.20
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However, Hollingshead et al. reported a major complication rate of 60% after
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thrombolysis through the superior mesenteric artery or portal vein, including one death
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due to procedure-related gastrointestinal hemorrhage.24 This indicates that thrombolysis
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should be reserved only for cases of very severe PVTs. To date, experience of
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transjugular intervention for the management of non-cirrhotic, non-malignant acute
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PVT is limited. One recent study reported the clinical outcomes of transjugular local
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thrombolysis with or without TIPS in 17 patients,25 showing a high rate of
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recanalization (94.1%), good 1 and 2 year PV patency rates (both 88.2%), and a major
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complication rate of 17.6%. Additional studies are required to clarify the clinical
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outcomes associated with this procedure.
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Prevention of PVT is of particular importance as it can be refractory (Table 1,
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patient 5) and fatal (Table 1, patient 11), despite the availability of effective therapeutic
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approaches. Although there was no consensus on this approach, prophylactic
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anticoagulation may be considered for patients with a high risk of PVT, including those
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with a hypercoagulable state, as seen in the present case. Indeed, prophylactic
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anticoagulation was applied in two case reports (Table 1, patients 4 and 10). Appropriate
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selection of the embolic materials is also important for the success of the procedure and
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prevention of PVT. Several embolic materials are available, including coils, glue,
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polyvinyl alcohol, microspheres, detachable balloons, and Amplatzer plugs.3,26-28
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However, coils or particles may migrate through the fistula thereby causing PVT,
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especially in large fistula.3 In this context, Landi et al. performed a successful
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transcatheter arterial embolization with N-butyl cyanoacrylate (NBCA) after occluding
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the draining vein using a balloon during the treatment of a mesenteric arteriovenous
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fistula, thereby minimizing migration of NBCA into the portal venous system.29 In conclusion, PVTs can occur after embolization of intrahepatic or extrahepatic
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APF and may result in devastating outcomes. Therefore, awareness and prevention of
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this potentially fatal complication are of great importance.
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References
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Ding P, Li Z, Han XW, Wang ZG, Zhang WG, Fu MT. Portal, mesenteric, and splenic vein thromboses after endovascular embolization for gastrointestinal
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Botelberge T, Van Vlierberghe H, Voet D, Defreyne L. Detachable balloon embolization of an arterioportal fistula following liver biopsy in a liver
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Table 1. Summary of demographics, clinical characteristics, embolization details, and outcomes of reported cases
Study
No./Age/Sex
Embolic
Prophylactic
Clinical
Cause of APF
embolization (artery-vein)
agent
[14]
2/62/F
Coils
No
4 days
Asymptomatic
SMV, MPV, LPV, RPV
Anticoagulation
Complete
SMA-SMV
Coils
No
3 days
SMV, MPV, LPV, RPV
Anticoagulation
Complete
Coils, glue
No
CHA, SMA, 3/36/M
Gunshot injury LGA-MPV
4/70/M
Unknown
GDA-SMV
Coils
Yes
Garg [5]
5/43/F
SB resection
SMA-SMV
AVP II
No
Hirakawa
Microwave 6/70/M
[11]
A3-LPV
Coils
No
coagulation
A7, A8-LPV
Microwave
A7-RPV,
coagulation
A8-RPV
9/60/F
Liver biopsy
Ding [4]
10/37/F
Zhao [15]
11/59/M
Coils
Hirakawa
Conservative
Complete
3 years
GI bleeding
SMV, MPV, RPV
TIPS
Partial
Abdominal
SMV, SV, MPV, LPV,
TIPS,
pain
RPV
thrombolysis
LPV, MPV, SMV
Anticoagulation
Complete
2 weeks
No
Hepatic
2 months encephalopathy
NA
Asymptomatic
RPV
Conservative
Partial
Coils, glue
No
NA
Asymptomatic
RPV
Anticoagulation
Partial
A7-RPV
Coils, glue
No
6 days
Asymptomatic
RPV
Anticoagulation
Partial
Unknown
SA-SV
Coils
Yes
10 days
Asymptomatic
SV, SMV, MPV
Anticoagulation
Complete
SB resection
SMA-SMV
Coils
No
2 days
Bil, Cr increase
SMV, MPV
Anticoagulation
No
8/70/F [11]
MPV
No
[11]
Hirakawa [11]
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Liver biopsy
Asymptomatic
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Hirakawa 7/63/F
2 days
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Diarrhea
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adhesiolysis
Chen [16]
Response
SMA-SMV SB resection Hysterectomy,
Mick [12]
Management
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Appendectomy, 1/62/M
Extent
manifestation
anticoagulation to PVT
Purow
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Interval from Fistula
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Present
12/65/F
Unknown
SA-SV
Coils, glue
No
9 days
Asymptomatic
SV, SMV, RPV
Anticoagulation
Partial
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Note: Response to anticoagulation/thrombolysis/conservative therapy was classified into “complete response” (no evidence of residual thrombus), “partial response” (a reduction of more than 50% of the thrombus), and “no response” in the other cases. APF, arterioportal fistula; PVT, portal vein thrombosis; SB, small bowel; SMA, superior mesenteria artery; SMV, superior mesesnteric vein; MPV, main portal vein; LPV, left portal vein; RPV, right portal vein; GDA, gastroduodenal artery; GI, gastrointestinal; TIPS, transjugular intrahepatic portosystemic shunt; SA, splenic artery; SV, splenic vein; AVP, Amplatzer vascular plug; Bil, bilirubin; Cr, creatinine.
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Figure legends
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Figure 1.
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Coronal reformatted image during the arterial phase of a CT scan, showing early
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opacification of an engorged splenic vein (arrow) and intrahepatic portal veins
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(arrowheads), suggesting a splenic arterioportal fistula (APF). Ascites due to high portal
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venous pressure and a hypoattenuating spleen indicating splenic infarction due to the
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steal phenomenon were also observed.
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Figure 2.
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(a) Arterial phase of digital subtraction arteriography (DSA) of the splenic artery just
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before embolization, showing the splenic artery (arrow) and rapid opacification of the
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hypertrophied splenic and portal vein (arrowheads). (b) Performance of complete
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splenic arterial embolization using multiple Interlock-18 detachable coils and a 1:2
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mixture of histoacryl and lipiodol.
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Figure 3.
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Coronal reformatted image obtained from a contrast-enhanced CT scan 9 days after
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embolization, showing the appearance of an extensive thrombosis involving the splenic
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artery (arrow) and portal vein (arrowheads).
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Figure 4.
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Coronal reformatted image obtained from a contrast-enhanced CT scan 2 months after
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embolization, showing a decrease in PVT volume. Also observed were new multifocal
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hypoattenuating lesions of the liver, indicating an ischemic change (arrows) and an
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inferior vena cava (IVC) filter (arrowhead) to prevent a pulmonary embolism from the
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deep vein thrombosis.
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S0890-5096(18)30585-5
DOI:
10.1016/j.avsg.2018.05.075
Reference:
AVSG 3984
To appear in:
Annals of Vascular Surgery
Received Date: 18 April 2018 Accepted Date: 1 May 2018
Please cite this article as: Kim PH, Gwon DI, Ko HK, Portal Vein Thrombosis after Endovascular Embolization of Splenic Artery for a Symptomatic Arterioportal Fistula: a Case report and Literature review, Annals of Vascular Surgery (2018), doi: 10.1016/j.avsg.2018.05.075. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Full Title Page
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Manuscript title: Portal Vein Thrombosis after Endovascular Embolization of Splenic
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Artery for a Symptomatic Arterioportal Fistula: a Case report and Literature review
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Manuscript type: Case report
Authors:
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Pyeong Hwa Kim
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Dong Il Gwon
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Heung Kyu Ko
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Department of Radiology, Asan Medical Center, College of Medicine, 88 Olympic-ro
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43-gil, Songpa-gu, Seoul 05505, Korea
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†
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Department of Radiology, Asan Medical Center, Ulsan University College of Medicine,
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88 Olympic-ro 43-gil, Songpa-gu, Seoul 05505, Korea
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Telephone: 82-2-3010-3974; Fax: 82-2-3010-6645; E-mail: [email protected]
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Corresponding author: Heung Kyu Ko
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E-mail list:
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Pyeong Hwa Kim: [email protected]
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Dong Il Gwon: [email protected]
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Heung Kyu Ko: [email protected]
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Acknowledgement: None.
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Disclosure statement:
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Funding: None.
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Conflict of Interest: The authors declare that they have no conflict of interest.
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Ethical approval: For this type of study formal consent is not required.
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Informed consent: The requirement to obtain written informed consent was waived from
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the IRB of Asan medical center.
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Portal Vein Thrombosis After Endovascular Embolization of the Splenic Artery for
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a Symptomatic Arterioportal Fistula: Case Report and Literature Review
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Abstract Arterioportal fistula (APF) can induce severe portal hypertension, and therefore
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requires appropriate and timely management. Endovascular treatment is increasingly
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used for the treatment of APFs due to its minimally invasive nature, although this
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procedure can lead to potentially fatal portal vein thrombosis (PVT). Reports of this
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complication are, however, rare. Here, we describe the case of a 65-year-old woman
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who experienced an extensive thrombosis from the splenic vein to the right portal vein
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following embolization of a splenic APF, leading to the requirement for ICU care and a
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prolonged hospital stay. In addition, the literature was reviewed to describe the clinical
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manifestations, diagnosis, and treatment of PVT after embolization of APF.
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Introduction Arterioportal fistulas (APFs), characterized by an abnormal communication
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between the systemic arteries and the portal vein, are rare but can be fatal. A systematic
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review of 88 patients with APFs reported the most frequent cause of APF to be trauma
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(28%), followed by iatrogenic causes (16%), congenital causes (15%), tumor (15%),
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and rupture of aneurysms (14%). Most APFs originated from the hepatic artery (65%),
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followed by the splenic (11%) and superior mesenteric arteries (10%).1 Because APFs
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can induce severe portal hypertension with multiple morbidities, appropriate and timely
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management is essential.
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Endovascular treatment is increasingly used for the treatment of intrahepatic
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and extrahepatic APFs because of low cost, repeatability, and minimally invasive
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nature.1-3 While some reports describe cases of extensive portal vein thrombosis (PVT)
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after embolization of mesenteric, splenic, or intrahepatic arteriovenous fistulas,4-6 this
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rare, but serious, complication remains poorly described.
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This report documents a case of PVT after the embolization of an APF and
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reviews the literature to identify common clinical manifestations of this condition, as
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well as its diagnosis and treatment.
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Case report This study was approved by the institutional review board (IRB); the
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requirement for informed consent was waived because of the retrospective nature of the
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case report. From January 1990 to October 2017, nine patients underwent embolization
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to treat an APF. Of those, one patient subsequently experienced a symptomatic PVT.
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A 65-year-old woman was referred to our institution due to progressive
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abdominal distension, dyspnea, and intermittent hematochezia during the previous
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month. At admission, a percutaneous peritoneal drainage catheter was inserted to drain
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ascites. The patient was alert, with a blood pressure of 144/92 mmHg and tachycardia
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(103 bpm). There was no history of heavy alcohol use. The only notable laboratory
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finding was an elevated serum creatinine level (3.74 mg/dL), necessitating dialysis.
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Other laboratory tests (including platelet count, prothrombin time, serum aspartate [AST]
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and alanine aminotransferases [ALT] concentration, bilirubin, alkaline phosphatase, and
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γ-glutamyl-transferase) were within the normal range. The patient was seronegative for
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hepatitis C viruses and positive for antibodies against hepatitis-B surface antigen.
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Contrast-enhanced computed tomography (CT) scan of the abdomen and pelvis
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showed the following: i) markedly dilated splenic vein with early opacification of the
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splenic and portal veins; ii) slightly dilated splenic artery; iii) homogeneously decreased
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attenuation of the spleen suggestive of splenic infarction; iv) a large amount of ascites;
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and v) mesenteric venous congestion suggestive of ischemic colitis (Figure 1). Surface
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nodularity of the liver, indicating liver cirrhosis, was not observed. Volume rendering
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images showed a direct communication between the splenic artery and vein, leading to a
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profuse inflow of blood to the liver and causing severe portal hypertension. A large
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volume of ascites was considered to be secondary to the portal hypertension. Acute
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history of medical renal disease and the patient’s kidneys and urinary tract were seen to
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be normal on CT scanning. Intermittent hematochezia was considered to be due to
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ischemic colitis, which was confirmed by colonoscopic biopsy.
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To isolate the splenic artery and portal venous system, angiographic
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management of the fistula was considered appropriate due to its minimally invasive
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nature. Selective splenic arteriography via the right femoral artery using a 5F Cobra II
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catheter (Cook Medical, Bloomington, IN) showed a fistula between the splenic artery
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and the markedly dilated splenic vein. The portal vein was not opacified in the venous
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phase of subsequent superior mesenteric arteriography. However, both direct
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embolization of the arteriovenous fistula and stent graft failed due to the strikingly high
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flow velocity, tortuosity of the splenic artery, and complex vascular anatomy. Because
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the spleen was totally infarcted, proximal splenic arterial embolization was therefore
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performed, using a 2.4F microcatheter (Boston Scientific Corp., Natick, MA) with
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multiple Interlock-18 detachable coils (Boston Scientific Corp.), one measuring 6 mm ×
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20 cm, two measuring 10 mm × 30 cm, and two measuring 20 mm × 50 cm. Histoacryl
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(B. Braun, Melsungen, Germany) was used in a 1:2 mixture with lipiodol. After
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completion of embolization, the fistula and splenic vein were not opacified on celiac
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arteriography and patent portal flow was observed during the venous phase of superior
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mesenteric arteriography, indicating successful occlusion of the splenic APF (Figure 2).
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After completion of the embolization procedure, the patient was closely
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observed in the intensive care unit. While the patient’s abdominal distension was
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initially relieved, distension recurred 1 week later. Follow-up CT 9 days after the
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embolization showed diffuse thrombosis of the splenic vein extending to the right
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ALT (24 to 56 U/L), alkaline phosphatase (98 to 245 IU/L), and total bilirubin (0.9 to
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4.4 mg/dL). At that time, creatinine was within normal range. The patient complained of
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pain in both legs, and duplex sonography showed extensive deep vein thrombosis (DVT)
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in the right femoral vein and the left posterior tibial vein. At that time, thrombocytosis
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(404,000/mL) was also noted. Since a hematologic disorder was suspected, bone
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marrow biopsy was performed, but the only remarkable finding was an increased
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number of megakaryocytes. No mutations were detected in JAK-2, MPL, and CALR
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gene studies. Therefore, the high platelet count was considered to be due to reactive
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thrombocytosis. To prevent the progression of PVT and DVT, heparin therapy was
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initiated (20,000 IU/day), and an IVC filter was inserted to prevent pulmonary
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thromboembolism. Follow-up CT 1 month after the embolization (2 weeks after
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initiating heparin therapy) showed a decreased PVT volume. However, discharge was
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delayed for a month due to the following comorbidities: i) recurrent episodes of fever
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caused by spontaneous bacterial peritonitis requiring antibiotic therapy and external
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drainage; ii) severe leg swelling and pain caused by DVT; and iii) post-AKI diuresis
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requiring hydration. Two months after embolization, CT scanning showed a further
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decrease in thrombus volume and no residual ascites (Figure 4). The patient was
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subsequently discharged with warfarin treatment. At the last follow-up (5 months after
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embolization), the patient remained clinically stable with no symptoms of portal
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hypertension. There were no recurrent episodes of hematochezia and abdominal
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distension. Creatinine levels were within normal range, and the extent of the DVT was
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further decreased. The patient’s platelet count remained elevated (510,000/mL), but this
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was considered likely to be reactive considering the unremarkable bone marrow biopsy
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results and infarcted spleen.
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Discussion APF is defined as an anomalous communication between the splanchnic artery
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and the portal venous system. It can be classified by location (intrahepatic or
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extrahepatic), or etiology (congenital or acquired). The most common cause of acquired
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APF is trauma (28%), primarily after a penetrating injury.7 Other causes include
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iatrogenic causes (16%), rupture of splanchnic aneurysms (14%), and, less frequently,
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cirrhosis and hepatic malignancy.1 In general, small and peripherally located APFs are
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asymptomatic and do not require therapeutic intervention. By contrast, large, centrally
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located APFs with high shunt flow are often symptomatic, inducing portal hypertension
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and requiring treatment.8 Despite the lack of data from large prospective studies,
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endovascular embolization is widely used for the treatment of symptomatic APFs
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because of its technical feasibility and good safety profile.6,9-11
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The patient described in this report experienced an extensive PVT following
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embolization of an APF with several related comorbidities. In this case, proximal
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splenic arterial embolization, rather than stent graft or direct embolization of the fistula,
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was performed. Although stent graft has the advantage of preserving arterial access, it is
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technically challenging due to the tortuous nature of the splenic artery, high flow
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velocity of the fistula, and absence of a distal landing zone.10,12 After the proximal
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splenic artery embolization, splenic venous flow became sluggish, inducing an
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extensive PVT. Right portal vein obliteration and multiple associated comorbidities
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necessitated a prolonged hospital stay (2 months). Therefore, greater awareness of this
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potentially fatal complication is required for better management and prevention.
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experienced PVT after embolization of APF (Table 1).4-6,13-17 Eight (66.7%) of these
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APFs were iatrogenic. The median interval from embolization to PVT diagnosis was 7.5
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days (range, 2 days to 3 years; interquartile range, 3.3–13 days). Reported clinical
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manifestations of PVT after embolization included abdominal pain, diarrhea, abrupt
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increases in bilirubin and creatinine levels, gastrointestinal bleeding, and hepatic
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encephalopathy. In 7 of the 12 patients, PVT was detected on follow-up imaging 2–10
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days after embolization, without clinical manifestation.
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In seven (87.5%) of eight patients, anticoagulation therapy alone was effective
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for treating PVTs. Anticoagulation therapy is a well-known, basic treatment for
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symptomatic PVT, recommended both by the American College of Chest Physicians
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(ACCP) and the American Association for the Study of Liver Disease (AASLD).18,19
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According to one systematic review,20 52.3% (116/228) of patients with PVT treated
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with anticoagulation had a complete (37.6%; 85/228) or partial (13.6%; 31/228)
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recanalization, with a low short-term complication rate of 1.3% (3/228, comprising
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minor retroperitoneal bleeding [n = 1], epistaxis [n = 1], and gum bleeding [n = 1]).
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Therefore, anticoagulation therapy can be an initial option with minimal safety concerns.
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Of note, a delay in initiating anticoagulation therapy is an independent predictor of
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recanalization failure.21 Therefore, prompt CT for the patients with recurred ascites,
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abdominal pain, or increased bilirubin and creatinine levels may be required for early
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detection of PVTs, thereby increasing the likelihood of successful recanalization.22,23
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Two patients (Table 1, patients 4 and 5) with extensive PVTs involving both the
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intrahepatic and extrahepatic portal vein required additional treatment, such as
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transjugular intrahepatic portosystemic shunt (TIPS) or thrombolysis. Thrombolysis is
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experienced a complete (40.8%; 29/71) or partial (45.1%; 32/71) recanalization.20
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However, Hollingshead et al. reported a major complication rate of 60% after
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thrombolysis through the superior mesenteric artery or portal vein, including one death
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due to procedure-related gastrointestinal hemorrhage.24 This indicates that thrombolysis
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should be reserved only for cases of very severe PVTs. To date, experience of
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transjugular intervention for the management of non-cirrhotic, non-malignant acute
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PVT is limited. One recent study reported the clinical outcomes of transjugular local
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thrombolysis with or without TIPS in 17 patients,25 showing a high rate of
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recanalization (94.1%), good 1 and 2 year PV patency rates (both 88.2%), and a major
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complication rate of 17.6%. Additional studies are required to clarify the clinical
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outcomes associated with this procedure.
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Prevention of PVT is of particular importance as it can be refractory (Table 1,
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patient 5) and fatal (Table 1, patient 11), despite the availability of effective therapeutic
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approaches. Although there was no consensus on this approach, prophylactic
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anticoagulation may be considered for patients with a high risk of PVT, including those
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with a hypercoagulable state, as seen in the present case. Indeed, prophylactic
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anticoagulation was applied in two case reports (Table 1, patients 4 and 10). Appropriate
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selection of the embolic materials is also important for the success of the procedure and
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prevention of PVT. Several embolic materials are available, including coils, glue,
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polyvinyl alcohol, microspheres, detachable balloons, and Amplatzer plugs.3,26-28
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However, coils or particles may migrate through the fistula thereby causing PVT,
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especially in large fistula.3 In this context, Landi et al. performed a successful
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transcatheter arterial embolization with N-butyl cyanoacrylate (NBCA) after occluding
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the draining vein using a balloon during the treatment of a mesenteric arteriovenous
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fistula, thereby minimizing migration of NBCA into the portal venous system.29 In conclusion, PVTs can occur after embolization of intrahepatic or extrahepatic
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APF and may result in devastating outcomes. Therefore, awareness and prevention of
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this potentially fatal complication are of great importance.
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References
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Table 1. Summary of demographics, clinical characteristics, embolization details, and outcomes of reported cases
Study
No./Age/Sex
Embolic
Prophylactic
Clinical
Cause of APF
embolization (artery-vein)
agent
[14]
2/62/F
Coils
No
4 days
Asymptomatic
SMV, MPV, LPV, RPV
Anticoagulation
Complete
SMA-SMV
Coils
No
3 days
SMV, MPV, LPV, RPV
Anticoagulation
Complete
Coils, glue
No
CHA, SMA, 3/36/M
Gunshot injury LGA-MPV
4/70/M
Unknown
GDA-SMV
Coils
Yes
Garg [5]
5/43/F
SB resection
SMA-SMV
AVP II
No
Hirakawa
Microwave 6/70/M
[11]
A3-LPV
Coils
No
coagulation
A7, A8-LPV
Microwave
A7-RPV,
coagulation
A8-RPV
9/60/F
Liver biopsy
Ding [4]
10/37/F
Zhao [15]
11/59/M
Coils
Hirakawa
Conservative
Complete
3 years
GI bleeding
SMV, MPV, RPV
TIPS
Partial
Abdominal
SMV, SV, MPV, LPV,
TIPS,
pain
RPV
thrombolysis
LPV, MPV, SMV
Anticoagulation
Complete
2 weeks
No
Hepatic
2 months encephalopathy
NA
Asymptomatic
RPV
Conservative
Partial
Coils, glue
No
NA
Asymptomatic
RPV
Anticoagulation
Partial
A7-RPV
Coils, glue
No
6 days
Asymptomatic
RPV
Anticoagulation
Partial
Unknown
SA-SV
Coils
Yes
10 days
Asymptomatic
SV, SMV, MPV
Anticoagulation
Complete
SB resection
SMA-SMV
Coils
No
2 days
Bil, Cr increase
SMV, MPV
Anticoagulation
No
8/70/F [11]
MPV
No
[11]
Hirakawa [11]
EP
Liver biopsy
Asymptomatic
AC C
Hirakawa 7/63/F
2 days
TE D
Nie [13]
Diarrhea
M AN U
adhesiolysis
Chen [16]
Response
SMA-SMV SB resection Hysterectomy,
Mick [12]
Management
SC
Appendectomy, 1/62/M
Extent
manifestation
anticoagulation to PVT
Purow
RI PT
Interval from Fistula
17
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Present
12/65/F
Unknown
SA-SV
Coils, glue
No
9 days
Asymptomatic
SV, SMV, RPV
Anticoagulation
Partial
RI PT
Note: Response to anticoagulation/thrombolysis/conservative therapy was classified into “complete response” (no evidence of residual thrombus), “partial response” (a reduction of more than 50% of the thrombus), and “no response” in the other cases. APF, arterioportal fistula; PVT, portal vein thrombosis; SB, small bowel; SMA, superior mesenteria artery; SMV, superior mesesnteric vein; MPV, main portal vein; LPV, left portal vein; RPV, right portal vein; GDA, gastroduodenal artery; GI, gastrointestinal; TIPS, transjugular intrahepatic portosystemic shunt; SA, splenic artery; SV, splenic vein; AVP, Amplatzer vascular plug; Bil, bilirubin; Cr, creatinine.
AC C
EP
TE D
M AN U
SC
315
18
ACCEPTED MANUSCRIPT 316
Figure legends
317
Figure 1.
319
Coronal reformatted image during the arterial phase of a CT scan, showing early
320
opacification of an engorged splenic vein (arrow) and intrahepatic portal veins
321
(arrowheads), suggesting a splenic arterioportal fistula (APF). Ascites due to high portal
322
venous pressure and a hypoattenuating spleen indicating splenic infarction due to the
323
steal phenomenon were also observed.
324
Figure 2.
325
(a) Arterial phase of digital subtraction arteriography (DSA) of the splenic artery just
326
before embolization, showing the splenic artery (arrow) and rapid opacification of the
327
hypertrophied splenic and portal vein (arrowheads). (b) Performance of complete
328
splenic arterial embolization using multiple Interlock-18 detachable coils and a 1:2
329
mixture of histoacryl and lipiodol.
330
Figure 3.
331
Coronal reformatted image obtained from a contrast-enhanced CT scan 9 days after
332
embolization, showing the appearance of an extensive thrombosis involving the splenic
333
artery (arrow) and portal vein (arrowheads).
334
Figure 4.
335
Coronal reformatted image obtained from a contrast-enhanced CT scan 2 months after
336
embolization, showing a decrease in PVT volume. Also observed were new multifocal
337
hypoattenuating lesions of the liver, indicating an ischemic change (arrows) and an
338
inferior vena cava (IVC) filter (arrowhead) to prevent a pulmonary embolism from the
339
deep vein thrombosis.
AC C
EP
TE D
M AN U
SC
RI PT
318
19
AC C
EP
TE D
M AN U
SC
RI PT
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AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT