Absence of portal vein bifurcation is a rare anomaly. We report a patient with this anomaly who underwent right hemihepatectomy for treatment of hepatocellular carcinoma. Although the procedure was carefully performed with a preoperative three-dimensional simulation and intraoperative cholangiography, postoperative portal vein thrombosis occurred

Arakawa, Yusuke

Ikemoto, Tetsuya

Imura, Satoru

Iwahashi, Shuichi

Morine, Yuji

Saito, Yu

Shimada, Mitsuo

Teraoku, Hiroki

Yamada, Shinichiro

Yoshikawa, Masato

Tokushima University Institutional Repository

INTRODUCTIONAbsence of portal vein bifurcation (APVB) is a rare anomaly thatwas initially reported in 1957 by Couinaud (1). Marks et al. re-ported that this anomaly, in which no left or right primary extra-hepatic branch exists, is present in 0.03%-2% of all cases (2). Failureto recognize this anomaly in patients receiving right hemihepatec-tomy could cause misligation of the main portal vein and portal veinthrombosis with multiple organ failure (3). Therefore, detailedpreoperative simulation and planning of liver resection are neces-sary (4). This report presents a rare case of APVB with postop-erative portal vein thrombosis following liver resection.CASE PRESENTATIONA 61-year-old man was referred to our department after a livertumor was detected at a health check-up. The patient had a historyof chronic hepatitis C and interferon therapy. Laboratory tests re-vealed the following : leukocytes, 5400/mm3 ; hemoglobin, 15.0g/dl ; platelets, 18.9104/mm3 ; albumin, 4.0 g/dl ; total bilirubin,0.7 mg/dl ; direct bilirubin, 0.1 mg/dl ; serum alanine transaminase,57 IU/l ; and prothrombin time, 10.5 s. The patient was negativefor hepatitis B surface antigen but positive for hepatitis C antibody,and hepatitis C viral RNA concentration was 4.0 log10 IU/mL. Theindocyanine green dye retention rate after 15 min was 9.1% (controlrange10%). Levels of serum tumor markers were as follows : α -fetoprotein, 3 ng/mL ; a proportion of α - fetoprotein L3 isoform,0.5% ; and des-γ -carboxy prothrombin, 1288 mAU/ml.Dynamic multi -detector computed tomography (MDCT) showeda contrast effect inside the tumor, which measured 10 cm in di-ameter, in the arterial phase (Fig. 1A). In the portal and delayedphases, the contrast agent was not retained. In addition, APVB wasdetected by MDCT (Fig.1B).The patient underwent right hemihepatectomy as a curativetreatment for hepatocellular carcinoma. In the hepatic hilum, theleft portal vein was not visible (Fig. 1C). Intraoperative ultrasonogra-phy was performed and hepatic transection was started along theCantlie line. Once the left portal vein was revealed in the transec-tion plane, the anterior and posterior Glissonean pedicles wereexposed (Fig. 1D). The optimal incision line was determined bypreoperative simulation with the SYNAPSE VINCENT volumeanalyzer (Fujifilm, Japan) (Fig. 2A). Intraoperative cholangiogra-phy was performed to confirm the resection line of the Glissoneanpedicles (Fig. 2B) before stapler -assisted resection (Fig. 2C andD). After the operation, ultrasonography showed normal intrahe-patic portal vein flow and no stenosis of the portal system.Until postoperative day 2, the patient’s course was uneventful.However, on postoperative day 3, ultrasonography revealed portalvein thrombosis with an increased volume of ascitic fluid in thedrainage tube. A portal thrombus was observed from the superiormesenteric vein through the left portal vein by CT (Fig. 3A). Ad-ministration of low-molecular -weight heparin and anti - thrombinformulation was initiated to dissolve the thrombus. The throm-bus did not increase in size, although CT showed the formation ofnew collateral vessels on postoperative day 8 (Fig. 3B). Ascitesdecreased gradually without any other complication, and the pa-tient was discharged from hospital on postoperative day 28.DISCUSSIONA normal branching pattern of the main portal vein into the rightand left portal veins is reported in80% of the population. The mostcommon anomaly is trifurcation of the main portal vein, which isobserved in10% of the population (5). In a previous report, APVBwas defined as follows (6). The extra-parenchymal left portal veinwas missing and the right portal vein entered the right hilum nor-mally. After that, the right portal vein formed right posterior andanterior branches and turned left towards the umbilical fissure.This anomaly must be detected in patients who have undergonehepatectomy. Unless this anomaly is detected preoperatively, thereis a risk of portal vein thrombosis resulting from wrongly identi-fying the portal vein branch and clamping the main portal vein.APVB in liverresection and postoperative fatal portal vein thrombo-sis were reported by Koh et al. in 1994 (3). After that report, casesCASE REPORTComplication of portal vein thrombosis after righthemihepatectomy in a patient lacking the portal veinbifurcationHiroki Teraoku, Yusuke Arakawa, Masato Yoshikawa, Shinichiro Yamada, Yu Saito, Shuichi Iwahashi,Tetsuya Ikemoto, Yuji Morine, Satoru Imura, and Mitsuo ShimadaDepartment of Digestive and Pediatric Surgery, Institute of Biomedical Sciences, Tokushima University Graduate School, Tokushima, JapanAbstract : Absence of portal vein bifurcation is a rare anomaly. We report a patient with this anomaly who un-derwent right hemihepatectomy for treatment of hepatocellular carcinoma. Although the procedure was care-fully performed with a preoperative three-dimensional simulation and intraoperative cholangiography, post-operative portal vein thrombosis occurred. J. Med. Invest. 63 : 315-318, August, 2016Keywords : portal vein anomaly, hepatectomy, portal vein thrombosisReceived for publication November 18, 2015 ; accepted June 26, 2016.Address correspondence and reprint requests to Hiroki Teraoku, MD,Institute of Biomedical Sciences, Department of Digestive and PediatricSurgery, Tokushima University Graduate School, 3 -18 -15 Kuramoto,Tokushima, 770-8503 Japan, and Fax : +81-88 -631-9698.The Journal of Medical Investigation Vol. 63 2016315Right dorsal branch Left branch Right anterior branch Right dorsal branch Left branch Right anterior branch Right dorsal branch Left branch Right anterior branch A B C D Post. A B C D left Ant. Anterior Glissonian pedicle  Posterior Glissonian pedicle  Fig 1. (A) Preoperative computed tomography imaging of the 10-cm tumor in the right hepatic lobe. (B) Maximum intensity projection imagingof the absence of portal vein bifurcation (yellow arrow) with an aberrant left branch originating from the right anterior branch. Intraoperative picture(C) prior to hepatic resection and (D) during hepatic resection showing the structure of the portal system (blue dotted lines).Fig 2. (A) Three-dimensional simulated imaging of the liver, tumor (yellow), hepatic artery (red), portal vein (pink), hepatic vein (blue) and inferiorvena cava (blue) using SYNAPSE VINCENT volume analyzer. Red dotted line shows the simulated line of parenchymal liver transection for righthepatectomy. (B) Intraoperative cholangiography after clamping the anterior (Ant.) and posterior (Post.) Glissonean pedicle. The resection lines(yellow dotted lines) were distant enough from the main tract, and the left branch was well contrasted. Intraoperative pictures before stapler -assistedresection of (C) anterior and (D) posterior Glissonean pedicles (white dotted lines).316 H. Teraoku, et al. PVT after hepatectomy with vein anomalyA B of APVB with safe hepatic resection for cancer treatment or safehepatic splitting for liver donation were reported (7, 8). Spampinatoet al. described major right hemihepatectomy in a patient withAPVB (4). They concluded that preoperative knowledge of thevascular structure from MDCT with three-dimensional recon-struction was beneficial for the surgeon to perform safe hepatec-tomy.In our case, APVB was recognized preoperatively ; however,portal vein thrombosis occurred. We performed intraoperativecholangiography and confirmed the presence of APVB by intraop-erative ultrasonography. However, the incision line for the rightanterior and posterior branches of the portal vein, using the stapler,may have been too close to the main portal vein, resulting in itslateral wall being cut.The Glissonean pedicle approach is a simple method of dissec-tion (9, 10). In cases of vascular anomaly, the ligation of the pedicleshould be performed near the liver parenchyma to avoid injuringthe elements that enter another section (9). Although we performedthe liver resection with care, the stapling line might be too closeto the right portal vein. Particularly in cases of APVB, the surgeonshould be aware of this danger ; careful Glissonean pedicle tran-section is required, or an intrafascial approach may be useful, whichopens Glisson’s capsule and treats the vessels individually.Portal vein thrombosis is a serious postoperative complicationof liver resection. Yoshiya et al. reported that the incidence of portalvein thrombosis after hepatectomy was 9.1%, and right hemihe-patectomy was a significant independent risk factor for main portalvein thrombosis (11). In the present case, the patient underwentright hemihepatectomy. Virchow’s triad of hypercoagulability,hemodynamic changes and endothelial injury are significant riskfactors for portal vein thrombosis, and it is reported that thesefactors usually coexist (12). A stapling line close to the right portalvein and the Pringle maneuver can affect the portal vein flow.The presence of ascites indicates insufficient portal flow causedby portal vein thrombosis (13). When portal vein thrombosis isfound, anticoagulation therapy or mechanical thrombectomy isrequired for early recanalization of the portal vein (13). In the pre-sent case, the portal vein thrombus was detected early by ultra-sonography, and anticoagulation therapy with low-molecular -weight heparin and anti - thrombin III was given. The thrombusremained ; however, sufficient collateral vessels were formed earlyto maintain the intrahepatic portal vein flow.APVB is a rare anomaly. Careful preoperative simulation andplanning is necessary. In the present case, portal vein thrombosisoccurred, although APVB was recognized preoperatively. Hence,meticulous care with Glissonean pedicle transection is requiredin patients with this anomaly.INFORMED CONSENTWritten informed consent was obtained from the patient for pub-lication of this case report and any accompanying images. A copyof the written consent is available for review by the Editor - in-Chiefof this journal.CONFLICT OF INTERESTThe authors declare that they have no conflict of interest.ACKNOWLEDGEMENTSWe thank Dr. Yoshimi Bando, a pathological faculty member, forthe initial pathological diagnosis of the patient in this case report.REFERENCES1. C C. Le foie : Etudes anatomiques et chirurgicales. Masson,Paris, 19572. Marks C : Developmental basis of the portal venous system.Am J Surg 117 : 671-81, 19693. Koh MK, Ahmad H, Watanapa P, Jalleh RP, Habib NA : Be-ware the anomalous portal vein. HPB Surg 7 : 237-9, 19944. Spampinato MG, Baldazzi G, Polacco M, Vigo M, del MedicoP, Gringeri E, Cillo U : Right hemihepatectomy in presenceof congenital absence of portal vein bifurcation : a challengingbut feasible procedure. J Am Coll Surg 215 : e1-4, 20125. Couinaud C : Surgical anatomy of the liver revisited. Couinaud,Paris, 19896. Chaib E : Absence of bifurcation of the portal vein. Surg RadiolAnat 31 : 389-92, 20097. Yamamoto H, Nagino M, Kawabata Y, Maki A, Takenouchi Y,Iwahashi N, Hayakawa N, Nimura Y : Resection of a hilar cho-langiocarcinoma in a patient with absent portal bifurcation.Hepatogastroenterology 47 : 362-4, 20008. Mitchell A, Mirza D, de Ville de Goyet J, Buckels J : Absenceof the left portal vein : a difficulty for reduction of liver grafts?Transplantation 69 : 1731-2, 20009. Yamamoto M, Katagiri S, Ariizumi S, Kotera Y, Takahashi Y :Fig 3. (A) CT on postoperative day 3. Thrombus of the portal vein was detected (arrowheads). (B) CT on postoperative day 8. Collateral vessels(yellow dotted circles) formed around the hepatoduodenal ligament. CT computed tomography.The Journal of Medical Investigation Vol. 63 August 2016 317Glissonean pedicle transection method for liver surgery (withvideo). J Hepatobiliary Pancreatic sci 19 : 3 -8, 201210. Yamamoto M, Katagiri S, Ariizumi S, Kotera Y, Takahashi Y,Egawa H : Tips for anatomical hepatectomy for hepatocellularcarcinoma by the Glissonean pedicle approach (with videos).J Hepatobiliary Pancreatic sci 21 : 53-6, 201411. Yoshiya S, Shirabe K, Nakagawara H, Soejima Y, Yoshizumi T,Ikegami T, Yamashita Y, Harimoto N, Nishie A, Yamanaka T,Maehara Y : Portal vein thrombosis after hepatectomy. WorldJ Surg 38 : 1491-7, 201412. Janssen HL, Wijnhoud A, Haagsma EB, van Uum SH, vanNieuwkerk CM, Adang RP, Chamuleau RA, van Hattum J,Vleggaar FP, Hansen BE, Rosendaal FR, van Hoek B : Extra-hepatic portal vein thrombosis : aetiology and determinantsof survival. Gut 49 : 720-4, 200113. Thomas RM, Ahmad SA : Management of acute post-operativeportal venous thrombosis. J Gastrointest Surg 14 : 570-7, 2010318 H. Teraoku, et al. PVT after hepatectomy with vein anomaly

Complication of portal vein thrombosis after right hemihepatectomy in a patient lacking the portal vein bifurcation

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Complication of portal vein thrombosis after right hemihepatectomy in a patient lacking the portal vein bifurcation

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