Biliary angiodysplasia is extremely rare. Our background search revealed only a few case reports in the English literature. We present a case of angiodysplasia of the proximal common bile duct in a patient with subacute upper gastrointestinal bleeding and symptomatic anemia. A standard esophagogastroduodenoscopy with subsequent dedicated duodenoscopy revealed blood-stained bile draining from the major ampulla orifice. A contrast-enhanced magnetic resonance cholangiopancreatography was unrevealing for any pancreaticobiliary pathology. The patient subsequently underwent an endoscopic retrograde cholangiopancreatography and SpyGlass® cholangioscopy, which demonstrated intermittent bleeding from angiodysplasia in the proximal common bile duct

Foong, Kap Sum

Kudakachira, Shaismy

Lee, Ashley

Ramberan, Hemchand

ACG Case Reports Journal

English

PubMed

Kap Sum Foong

Ashley Lee

Shaismy Kudakachira

Hemchand Ramberan

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Hemobilia from Biliary Angiodysplasia Diagnosed with Cholangioscopy

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Washington University School of MedicineDigital Commons@BeckerOpen Access Publications2016Hemobilia from biliary angiodysplasia diagnosedwith cholangioscopyKap Sum FoongCrozer Chester Medical CenterAshley LeeWashington University School of Medicine in St. LouisShaismy KudakachiraCrozer Chester Medical CenterHemchand RamberanCrozer Chester Medical CenterFollow this and additional works at: https://digitalcommons.wustl.edu/open_access_pubsThis Open Access Publication is brought to you for free and open access by Digital Commons@Becker. It has been accepted for inclusion in OpenAccess Publications by an authorized administrator of Digital Commons@Becker. For more information, please contact engeszer@wustl.edu.Recommended CitationFoong, Kap Sum; Lee, Ashley; Kudakachira, Shaismy; and Ramberan, Hemchand, ,"Hemobilia from biliary angiodysplasia diagnosedwith cholangioscopy." ACG Case Reports Journal.3,4. e132. (2016).https://digitalcommons.wustl.edu/open_access_pubs/5706CASE REPORT | BILIARYHemobilia from Biliary Angiodysplasia Diagnosed withCholangioscopyKap Sum Foong, MD1, Ashley Lee, MD2, Shaismy Kudakachira, DO1, andHemchand Ramberan, MD31Department of Medicine, Crozer Chester Medical Center, Chester, PA2Department of Radiology, Washington University, St. Louis, MO3Division of Gastroenterology, Crozer Chester Medical Center, Chester, PAABSTRACTBiliary angiodysplasia is extremely rare. Our background search revealed only a few case reports in theEnglish literature. We present a case of angiodysplasia of the proximal common bile duct in a patient withsubacute upper gastrointestinal bleeding and symptomatic anemia. A standard esophagogastroduodeno-scopy with subsequent dedicated duodenoscopy revealed blood-stained bile draining from the majorampulla oriﬁce. A contrast-enhanced magnetic resonance cholangiopancreatography was unrevealing for anypancreaticobiliary pathology. The patient subsequently underwent an endoscopic retrograde cholangiopan-creatography and SpyGlass® cholangioscopy, which demonstrated intermittent bleeding from angiodysplasiain the proximal common bile duct.INTRODUCTIONAngiodysplasia of the gastrointestinal tract is one of the common causes of occult gastrointestinal tract bleeding.However, angiodysplasia of the common bile duct (CBD) is extremely rare. Our literature review revealed only afew reported cases of biliary angiodysplasia.1-3CASE REPORTA 75-year-old male with coronary artery disease and indwelling coronary stents on dual antiplatelet therapy withclopridogel and aspirin for 5 years presented with chest pain, progressive dyspnea, and fatigue for a week. He alsoendorsed passing black tarry stools intermittently for the last 6 months. The patient had no history of epistaxis,chronic kidney disease, aortic stenosis, vonWillebrand’s disease, or hereditary hemorrhagic telangiectasia.The physical examination was unremarkable except for pallor. There was no mucocutaneous telangiectasia. Stoolguaiac test was positive. The initial laboratory tests were signiﬁcant for an iron deﬁciency anemia with a hemoglo-bin of 7.6 g/dL (baseline, 12-15 g/dL). Complete metabolic panel was within normal limits. The patient’s clinical pre-sentation and supporting laboratory data were suspicious for subacute and probable insidious uppergastrointestinal bleeding. Both of his antiplatelet agents were immediately discontinued. Proton pump inhibitortherapy was initiated. He had an appropriate increase of his hemoglobin after 2 U of blood transfusion.A diagnostic esophagogastroduodenoscopy was performed within 24 hours of admission, which demonstrateddark red blood around the major ampulla on limited tangential views. There was no evidence of varices, ulcer dis-ease, or other mucosal vascular anomalies to the examined third portion of the duodenum. A dedicated duodenos-copy with en-face view of the major ampulla showed blood-stained bile and dark red blood emanating from theACG Case Rep J 2016;3(4):e132. doi:10.14309/crj.2016.105. Published online: September 28, 2016.Correspondence: Kap Sum Foong, Department of Medicine, Crozer Chester Medical Center, 1 Medical Center Blvd, Upland, PA 19013(KapSum.Foong@crozer.org).Copyright: © 2016 Sum Foong et al. This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0International License. To view a copy of this license, visit http://creativecommons.org/licenses/by-nc-nd/4.0.ACG Case Reports Journal / Volume 3 / Issue 4 acgcasereports.gi.org 1ACGCASE REPORTS JOURNALmajor ampulla, concerning for hemobilia or hemosuccus pan-creaticus (Figure 1). There was no evidence of mass lesion. Afollow-up contrast-enhanced magnetic resonance cholan-giopancreatography was unrevealing for any mass lesionsor intraductal ﬁlling defects. Endoscopic retrograde chol-angiopancreatography and SpyGlass® (Boston Scientiﬁc,Marlborough, MA) cholangioscopy were subsequently pur-sued, which showed blood-stained bile during the initialinspection. After thorough irrigation and suction, clear vis-ualization of the area showed diminutive arborizing andectatic blood vessels with intermittent blood oozing in theproximal CBD, consistent with angiodysplasia (Figure 2).The size of the lesion was estimated to be less than 3.3mm. Selective cannulation and inspection of the left andright main hepatic ducts, its immediate radicles, and thecystic duct did not reveal any bleeding source or similarappearing angiodysplasia. No endoscopic therapy was per-formed to the lesion as SpyGlass® compatible accessoriesfor intraductal bleeding treatment were unavailable. Dualantiplatelets remained discontinued after the procedure.The patient’s chest pain andmelena resolved during his hospi-talization. His hemoglobin level remained stable at 10-10.5g/dL. In light of stable hemoglobin and no evidence of recur-rent bleeding, no further angiography or medical treatmentwas pursued. Dual antiplatetet therapy was not restarted ondischarge. Outpatient follow-up at 1 and 3 months revealedhemoglobin of 12.2 and 12.0 g/dL, respectively. The patientlater underwent an outpatient colonoscopy and terminal ileo-scopy, which were negative for bleeding and mucosalpathology.DISCUSSIONBiliary angiodysplasia is an extremely rare clinical entity ofhemobilia. Although the pathogenesis of biliary angiodyspla-sia is not well understood, it could be similar to gallbladderand colonic angiodysplasia.4-6 The contraction and relaxationof the CBD with resultant intermittent occlusion of the ves-sels may lead to focal ectatic dilation of the mucosal vessels.Biliary angiodysplasia can be seen in patients with hereditaryhemorrhagic telangiectasia.1,2Identifying the source of hemobilia is crucial to preventpotential complications. In the era of advanced endoscopictechnique, cholangioscopy allows direct visualization of thebiliary system to determine the source of hemobilia.1,2,3,7-11Aydinli et al described the intraoperative application of chol-angioscopy to determine the source of hemobilia in selectedpatient in order to guide surgical intervention.7 A study byGreen et al suggests that patients who are diagnosedadequately based on endoscopic and cholangioscopic ﬁnd-ings do not require more deﬁnitive investigations.9Current available chalangioscopy includes SpyGlass® chol-angioscopy and direct peroral cholangioscopy (DPC).SpyGlass® cholangioscopy is a 3.3-mm 4-lumen catheterwith a 1.2-mm accessory channel. Its small size enables bet-ter access to biliary tree at the cost of image quality. Inaddition, it has limited endoscopic therapy secondary toFigure 1. (A) Blood-stained bile pooling near to the major ampulla oriﬁce.(B) Duodenoscopy with en-face view showed hemobilia with dark reddishblood emanating from the major ampulla oriﬁce.Figure 2. (A) SpyGlass® cholangioscopy revealed ectatic blood vessels in the proximal CBD after thorough irrigation and suction. (B and C) Close upviews of the angiodysplasia.Sum Foong et al Hemobilia from Angiodysplasia of Common Bile DuctACG Case Reports Journal / Volume 3 / Issue 4 acgcasereports.gi.org 2small accessory channel.10,11 DPC is an ultraslim endoscopewith an outer diameter of 5-6 mm. It has a larger workingchannel enabling the accommodation of argon plasmacoagulation probes, larger biopsy forceps, and lithotripsyﬁbers. It also has superior image quality. However, DPCrequires a larger sphincterotomy and dilated bile duct.12 Italso lacks of access to proximal common bile duct.Therefore, the proximal biliary angiodysplasia in our patientwould have not been identiﬁed had we used DPC.Transarterial embolization is the ﬁrst line of interventionto stop bleeding from hemobilia.9 Hayashi et al andKomaki et al reported that biliary angiodysplasia could betreated with coil embolization and arterial embolization,respectively.1,2 Literature revealed successful neodymium-doped yttrium aluminum garnet (Nd-YAG) laser coagula-tion and argon plasma coagulation to control bleedingfrom biliary angiodysplasia using DPC.2,3 The presentpatient was managed successfully with discontinuation ofdual antiplatelet therapy. Given the unexpected locationof the biliary angiodysplasia and the lack of availableSpyGlass® cholangioscopy compatible therapeutic accesso-ries, no endoscopic intervention for bleeding was per-formed. Some studies showed the association betweenantiplatelet therapy use and increased risks of morbidityand mortality from gastrointestinal bleeding.13 Cessation ofantiplatelet therapy signiﬁcantly lowered our patient’s riskof rebleeding.DISCLOSURESAuthor contributions: KS Foong interpreted the data, wrotethe manuscript, and is the article guarantor. A. Lee criticallyrevised the manuscript. S. Kudakachira collected the data. H.Ramberan contributed to the intellectual content of this caseand approved the ﬁnal manuscript.Financial disclosure: None to report.Informed consent was obtained for this case report.ReceivedNovember 15, 2015;AcceptedMarch 11, 2016REFERENCES1. Hayashi S, Baba Y, Ueno K, et al. Small arteriovenous malformation ofthe common bile duct causing hemobilia in a patient with hereditaryhemorrhagic telangiectasia. Cardiovasc Intervent Radiol. 2008;31(Suppl2):S131–4.2. Komaki Y, Kanmura S, Funakawa K, et al. A case of hereditary hemor-rhagic telangiectasia with repeated hemobilia arrested by argon plasmacoagulation under direct peroral cholangioscopy. Gastrointest Endosc.2014;80(3):528–9.3. Siddique I, Galati J, Ankoma-Sey V, et al. The role of choledochoscopy inthe diagnosis and management of biliary tract diseases. GastrointestEndosc. 1999;50(1):67–73.4. Yudt WM, Silverman ED, Kistler AM. Scintigraphic detection of hemo-bilia complicating angiodysplasia. J Nucl Med. 1994;35(5):870–1.5. Kok KYY, Telisinghe PU. Angiodysplasia of the gallbladder. Int J SurgCase Rep. 2011;2(8):256–7.6. Boley SJ, Sammartano R, Adams A, et al. On the nature and etiology ofvascular ectasias of the colon. Degenerative lesions of aging.Gastroenterology. 1977;72:650–60.7. Aydinli M, Koruk I, Koruk S, et al. Intraoperative cholangioscopy with anultrathin endoscope for hemobilia. Endoscopy. 2011;43(Suppl 2 UCTN):E410.8. Moon JH, Choi HJ, Ko BM. Therapeutic role of direct peroral cholangio-scopy using an ultra-slim upper endoscope. J Hepatobiliary PancreatSci. 2011;18(3):350–6.9. Green MH, Duell RM, Johnson CD, et al. Haemobilia. Br J Surg. 2001;88(6):773–86.10. Williamson JB, Draganov PV. The usefulness of SpyGlassTM choledo-choscopy in the diagnosis and treatment of biliary disorders. CurrGastroenterol Rep. 2012;14(6):534–41.11. Parsi MA. Peroral cholangioscopy in the new millennium. World JGastroenterol. 2011;17(1):1–6.12. Monga A, Ramchandani M, Nageshwar Reddy D. Per-oral cholangio-scopy. J Interv Gastroenterol. 2011;1(2):70–7.13. Alli O, Smith C, Hoffman M, et al. Incidence, predictors, and outcomesof gastrointestinal bleeding in patients on dual antiplatelet therapy withaspirin and clopidogrel. J Clin Gastroenterol. 2011;45:410–4.Sum Foong et al Hemobilia from Angiodysplasia of Common Bile DuctACG Case Reports Journal / Volume 3 / Issue 4 acgcasereports.gi.org 3

Hemobilia from biliary angiodysplasia diagnosed with cholangioscopy

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Hemobilia from biliary angiodysplasia diagnosed with cholangioscopy

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