Successful Treatment of Bleeding Rectal Varices with Balloon-Occluded Retrograde Transvenous Obliteration in a Patient with a History of Sigmoidectomy

Case Report

1) Department of Diagnostic Radiology, Fukuiken Saiseikai Hospital, Japan 2) Department of Gastroenterology, Fukuiken Saiseikai Hospital, Japan

Keiichi Nagai1), Shiro Miyayama1), Masashi Yamashiro1), Jun Tohyama1), Kenshi Kawamura1), Taku Sanada2)

Abstract Rectal varices most commonly develop in patients with portal hypertension due to liver cirrhosis. Although rectal varices rarely cause bleeding, their rupture can lead to life-threatening massive hemorrhages. Balloon- occluded retrograde obliteration (BRTO) is feasible in cases of varices involving a single or a few drainage vessels. Although rectal varices usually have numerous drainage vessels, the present case involved a single drainage vessel. The unusual presentation of the current case was considered to have been caused by a previous surgery for sigmoid colon carcinoma. BRTO was successfully performed through the left internal iliac vein, achieving complete hemostasis of the rectal varices. Key words: Rectal varices, Balloon-occluded retrograde obliteration (BRTO), venous intervention, portal hypertention, cirrhosis (Interventional Radiology 2016; 1: 19-22)

owing to their complex hemodynamics. Introduction In this paper, we present a case of a patient with bleeding rectal varices successfully treated with BRTO through a sin- The development of portosystemic collateral vessels is gle drainage vein (the left internal iliac vein). We also dis- common in portal hypertension due to liver cirrhosis [1]. cuss the treatment options for rectal varices based on their Rectal varices are a type of ectopic varices that occur out- vascular anatomy. side of the gastroesophageal region. Bleeding from rectal varices is rare; however, it has the potential to cause massive Case Report life-threatening hemorrhage [2]. Various treatments, such as surgical or endoscopic liga- A 64-year-old woman with a recurrent tumor (sigmoid tion, endoscopic injection sclerotherapy (EIS), percutaneous colon carcinoma) in her pelvic cavity was admitted to our transhepatic obliteration (PTO) with modified techniques, hospital. She had undergone resection for sigmoid colon car- transjugular intrahepatic portosystemic shunt (TIPS), and cinoma 4 years prior. She also had liver cirrhosis related to balloon-occluded retrograde transvenous obliteration a hepatitis C virus infection and hepatocellular carcinoma (BRTO), have been used to control bleeding from rectal (HCC). She had undergone multiple treatments, including varices; however, there is no standard treatment for the con- partial hepatectomy, radiotherapy, and radiofrequency abla- dition [2-10]. In general, rectal varices have numerous drain- tion for HCC during the last 3 years. Radiotherapy was per- age vessels that pass through the pelvic venous plexus; formed for the treatment of recurrent sigmoid colon carci- therefore, BRTO is usually not feasible for rectal varices, noma due to poor hepatic functional reserve (Child-Pugh

Received: October 19, 2015. Accepted: February 3, 2016. Correspondence Author: Keiichi Nagai. E-mail: [email protected]

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A B

Figure 1. Preoperative computed tomography (CT) images. A. Contrast-enhanced CT image showing large tortuous rectal varices protruding into the rectal lumen (arrow). B. The superior mesenteric vein is identified as the feeding vessel (arrow), and the left internal iliac vein is considered to be the only drainage vessel (arrowhead). class C; score: 11). The laboratory tests showed the follow- ing results: hemoglobin, 11 g/dL; platelet, 87,000/μL; total bilirubin, 3.5 mg/dL; serum albumin, 2.4 g/dL; and prothrombin time, 61.5%. Three weeks after the start of radiotherapy, she experi- enced transient anorectal bleeding twice during the periods of radiation therapy. The contrast-enhanced computed tomography (CT) scan showed large tortuous rectal varices fed by the superior mesenteric vein and draining into the left internal iliac vein (Fig.1). Three days later, a massive bleeding occurred, and the patient became hemodynamically unstable. Laboratory data showed severe anemia with 7.5 g/ dL serum hemoglobin. Emergency endoscopy was performed, and bleeding from large rectal varices was detected (Fig.2). Although EIS with a mixture of n -butyl-2- cyanoacrylate (histoacryl; B. Braun, Melsungen, Germany) Figure 2. Endoscopic images. Bleeding from giant rectal and iodized oil (lipiodol; Andre Guerbet, Aulnay-sous-Bois, varices can be seen from the images. France) achieved temporary hemostasis, it was considered that further bleeding could occur. Emergent TIPS creation or PTO might be a therapy of choice; however, these were con- The catheter and sheath were withdrawn, and a 25-cm-long traindicated because of marked dilatation of the bile ducts in 7-F sheath that had been steam-shaped into a J-curve was the left hepatic lobe due to the previous treatment for HCC advanced into the left internal iliac vein over the wire. Then, and multiple peribiliary cysts in the right hepatic lobe, as a 6-F balloon catheter (13 mm balloon diameter) (Selecon well as a small amount of ascites. The trans-ileal approach MP Catheter II; Terumo Clinical Supply, Kakamigahara, Ja- might also be difficult because of adhesion after abdominal pan) was advanced into the left internal iliac vein. Balloon- surgery. On the basis of this anatomical information, we occluded venography of the left internal iliac vein showed considered that it might be possible to perform BRTO retrograde opacification of the rectal varices, as well as of through the left internal iliac vein. Written informed consent the superior and inferior gluteal veins. After embolization of was obtained from the patient, and she was transferred to the gluteal veins with microcoils (Nester embolization coil; the angiography room. Cook, Bloomington, IN, USA) through a microcatheter Under local anesthesia, a 5-F sheath was first inserted (Progreat β3, Terumo Clinical Supply), stepwise infusion of through the right femoral vein. A 0.035-inch hydrophilic a sclerosant, 5% EOI (a mixture of 10% ethanolamine oleate guidewire (Radifocus M; Terumo, Tokyo, Japan) was then [Oldamin; Takeda, Osaka, Japan] and the same amount of advanced into the left internal iliac vein through a 5-F shep- iopamidol [300 mg I/mL Iopamiron; Bayer, Leverkusen, herd’s hook catheter placed in the left common iliac vein. Germany]), was performed at intervals of 10-30 min through

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Figure 3. Spot radiograph images. The spot radiograph obtained during the procedure of balloon-occluded retrograde Figure 4. Postoperative computed tomographic (CT) images. obliteration shows the complete filling of the rectal varices The contrast-enhanced CT scan obtained 2 weeks after the with 5% ethanolamine oleate with iopamidol, and opacifica- balloon-occluded retrograde obliteration procedure shows that tion of the other drainage vessels of the right internal iliac vein the rectal varices had been completely obliterated (arrow). (arrowheads). The arrow indicates the metallic coils in the left gluteal vein. veins of the inferior mesenteric system and the middle and inferior rectal veins of the iliac system [1]. a balloon catheter under balloon occlusion. The infusion The most common cause of rectal varices is portal hyper- procedure was ended when the varices had been filled with tension due to liver cirrhosis, which is present in 3.6-78% of EOI, and the other collateral vessels of the right internal all cases [2, 3]. Although the clinical incidence of bleeding iliac vein were visualized (Fig.3). The total volume of in- in patients with rectal varices is 0.5-3.6%, the rupture of fused 5% EOI was 38 mL (stepwise injection of 20, 10, and rectal varices can lead to massive and life-threatening hem- 8 mL, respectively), and the balloon occlusion period lasted orrhage [2]. for 70 min. After aspiration of the residual EOI and clots in The standard treatment for rectal varices is EIS and/or en- the vessel lumen through the balloon catheter, the catheter doscopic variceal ligation [3]. However, when these endo- and sheath were withdrawn. During the procedure, 4000 U scopic treatments are not sufficient to achieve hemostasis, human haptoglobin (Benesis, Osaka, Japan) was adminis- especially in cases of large varices or massive bleeding, ad- tered intravenously to prevent renal dysfunction. Although ditional therapy might be necessary. Surgical ligation is an 38 mL of EOI was a relatively large volume compared with effective treatment for bleeding anorectal varices [4]. How- the patient’s body weight (58 kg), no renal dysfunction de- ever, it is not recommended for patients with massive bleed- veloped after BRTO. ing and/or exhibit severe liver dysfunction. TIPS is also an After the BRTO procedure, no further bleeding occurred, effective treatment for varices caused by portal hypertension and the patient’s hemoglobin level stabilized at 11.5 g/dL [5-7]. In a report by Vidal et al. [7] on TIPS for the control and the hepatic functional reserve had not been changed. of ectopic variceal bleeding, hemostasis was achieved in all Contrast-enhanced CT scan obtained 2 weeks after the pro- 24 cirrhotic patients. Rebleeding occurred in six patients, cedure revealed complete obliteration of the rectal varices and the cumulative rebleeding rate at 1 and 2 years was (Fig.4). On CT obtained 5 months after BRTO, ascites 23% and 31%, respectively. PTO can also be used to di- slightly increased; however, recurrence of the rectal varices rectly embolize rectal varices. In this procedure, obliteration and aggravation of other varices were not observed. To date, of the inflow vessel alone enables the eradication of rectal recurrent rectal bleeding has not developed during the 6- varices, even if they have numerous outflow veins. Addition- month follow-up period. ally, some modifications, such as coil embolization or antegrade balloon occlusion, can enhance the therapeutic effect Discussion due to prolonging the retention time of a sclerosant in the varices [8]. However, this procedure is generally contraindi- Rectal varices represent portal systemic collaterals that cated for patients with ascites. The trans-ileal approach or manifest as discrete dilated submucosal veins and constitute direct puncturing of the paraumbilical vein is required to ac- a pathway for portal venous flow between the superior rectal cess rectal varices in such patients [9].

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BRTO is an established procedure for the management of gastric varices in Japan, and has shown promising results in References the past decade. The BRTO technique is efficient, especially 1. Sharma M, Rameshbabu CS. Collateral pathways in portal hyper- in cases of varices involving a single or a few drainage ves- tension. J Clin Exp Hepatol 2012; 2(4): 338-352. doi: 10.1016/j. sels. The superior rectal venous plexus normally drains into jceh.2012.08.001. Epub 2012 Dec 16. the portal vein through the inferior mesenteric vein. As por- 2. Yoshino K, Imai Y, Nakazawa M, Chikayama T, Ando S, Suga- wara K, et al. Therapeutic strategy for patients with bleeding rectal pressure increases, the superior rectal vein changes from tal varices complicating liver cirrhosis. Hepatol Res 2014; 44(11): an outflow vessel to an inflow vessel, and drains through the 1088-1094. doi: 10.1111/hepr.12232. Epub 2014 Jan 7. middle and inferior rectal veins into numerous internal iliac 3. Watanabe N, Toyonaga A, Kojima S, Takashimizu S, Oho K, branch veins. Although some previous reports have de- Kokubu S, et al. Current status of ectopic varices in Japan: results scribed the use of BRTO to treat rectal varices [2,10], it is of a survey by the Japan Society for Portal Hypertension. Hepatol not usually indicated for rectal varices because rectal varices Res 2010; 40(8):763-776. doi: 10.1111/j.1872-034X.2010.00690.x. usually have multiple drainage veins, and numerous collater- 4. Hosking SW, Johnson AG. Bleeding anorectal varices―a misun- als between the iliac venous branches are often difficult to derstood condition. Surgery 1988; 104(1):70-73. completely occlude with a balloon catheter. In the present 5. Katz JA, Rubin RA, CopeC, Holland G, Brass CA. Recurrent case, the superior rectal vein might have been ligated during bleeding from anorectal varices: successful treatment with a the previous sigmoidectomy procedure, and the hemodynam- transjugular intrahepatic portosystemic shunt. Am J Gastroenterol. ics of the pelvic venous plexus might also have changed. As 1993; 88(7):1104-1107. 6. a result, rectal varices that were drained by a single blood Ahn SS, Kim EH, Kim MD, Lee WJ , Kim SU. Successful hemostasis of intractable rectal variceal bleeding using variceal emboli- vessel (the left internal iliac vein) developed. On the basis zation. World J Gastroenterol 2015; 21(8): 2558-2562. doi: of this specific hemodynamics, BRTO was performed suc- 10.3748/wjg.v21.i8.2558. cessfully, and complete hemostasis of the rectal varices was 7. Vidal V, Joly L, Perreault P, Bouchard L, Lafortune M, Pomier- achieved. BRTO is considered a less invasive treatment than Layrargues G. Usefulness of transjugular intrahepatic portosys- other interventional procedures such as TIPS and PTO. In temic shunt in the management of bleeding ectopic varices in cir- cases of rectal varices in which a single drainage vessel is rhotic patients. Cardiovasc Intervent Radiol 2006; 29(2): 216-219. identified on CT, BRTO should be attempted first. Although 8. Minamiguchi H, Kawai N, Sato M, Ikoma A, Sanda H, Nakata K, it is ideal that rectal varices be treated with BRTO alone, the et al. Successful treatment of endoscopically unmanageable rectal risk of intraprocedural rebleeding during occlusion of the varices by balloon-occluded antegrade transvenous sclerotherapy outflow should not be ignored. Therefore, a combined tech- followed by microcoil embolization. J Vasc Interv Radiol 2013; 24 nique of BRTO and PTO/TIPS should be considered as an (9): 1399-1403. doi: 10.1016/j.jvir.2013.04.015. 9. Ibukuro K, Kojima K, Kigawa I, Tanaka R, Fukuda H, Abe S, et additional option, if possible, especially in high-flow or rup- al. Embolization of rectal varices via a paraumbilical vein with an tured cases. abdominal approach in a patient with massive ascites. J Vasc In- In summary, BRTO is an effective treatment for cases of terv Radiol 2009 ; 20 ( 9 ) : 1259-1261. doi : 10.1016 / j. rectal varices involving a single drainage vein, particularly jvir.2009.05.041. in patients who had undergone surgical reconstruction in the 10. Watanabe K, Imai Y, Takaya H, Nakazawa M, Chikayama T, Ando pelvic cavity. S, et al. A case of liver cirrhosis due to hepatitis C virus infection complicating giant anorectal varices treated with balloon-occluded Conflict of interest: The authors declare that they have no con- retrograde transvenous obliteration. Clin J Gastroenterol 2011; 4 flicts of interest to report. (1): 19-23. doi: 10.1007/s12328-010-0187-4. Epub 2010 Nov 25.