Letter to Editor

Bugbee flexible electrocautery facilitates choledochoscopic biopsy,fulguration,and debulking of a high grade intraductal papillary neoplasm of the bile duct

To the Editor:

1.4 统计学方法 采用SPSS 17.0统计软件进行分析，计量资料以表示，组间均数比较采用t检验，以P<0.05为差异有统计学意义。

Intraductal papillary neoplasm of the bile duct(IPNB)may occur in the setting of primary sclerosing cholangitis,choledochal cysts or hepatolithiasis[1].The entity may be a precursor of cancer or intrahepatic cholangiocarcinoma.Invasive carcinoma,tubular or mucinous adenocarcinoma is present in approximately 40%−80%of IPNBs[1–3].IPNB is frequently found in the resection margins of patients undergoing hepatectomy for cholangiocarcinoma[4].This report describes a patient with obstructive jaundice secondary to IPNB who underwent interventional radiology-operated choledochoscopic-guided flexible electrocautery and fulguration to facilitate diagnostic biopsy and debulking of intrabiliary tumor.

Institutional review board approval was obtained for this report.A 35-year-old woman presented with scleral icterus and severe pruritus.Contrast-enhanced computed tomography(CT)showed severe biliary ductal dilatation,multiple large intra-biliary filling defects(Fig.1),and bilateral cavitary lung nodules.Endoscopic retrograde cholangiopancreatography(ERCP)at an outside hospital was unsuccessful.A 10-French external biliary drain was placed to provide drainage of the biliary system.However,the catheter could not penetrate the obstructed segment.Two weeks later,the patient presented with persistent jaundice,bilirubin of 16.6 mg/dL(normal:＜1.2 mg/dL),alkaline phosphatase of 527 IU/L(normal:30–116 IU/L),and CA 19–9 of 3029 U/mL(normal:0–40 U/mL).Interventional radiology was consulted to provide direct biliary visualization,obtain additional biopsies,and attempt internal/external biliary drain placement.

旅游已经逐渐地深入到我国人民的日常生活中，高校作为对旅游人才进行培养的重要基地，应该对旅游行业的变化进行积极关注，以培养出适合旅游市场发展的专业人才，从而对我国旅游业的发展起到促进作用。

Fig.1.Axial computed tomography image demonstrates diffuse intrahepatic biliary ductal dilatation(arrowheads)and a large mass(arrow)within the central biliary tree.Numerous additional filling defects in right and left sided bile ducts representing masses were also seen within the biliary tree,not pictured.

Fig.2.Fluoroscopic image during contrast injection through the patient’s partially retracted existing biliary drain demonstrates diffuse ductal dilatation of the intrahepatic bile ducts(arrow)but notably absent filling of the extrahepatic bile ducts.Obstruction was traversed with a 4-French Cobra Glidecath(Terumo Interventional Systems)and Glidewire(Terumo Interventional Systems).

Fig.3.(A)A 16.5-French flexible endoscope(Olympus Medical)is seen within the biliary tree(arrow).The 5-French Bugbee fulgurating electrode(Gyrus ACMI)is seen exiting the working channel of the endoscope(arrowhead).Note is made of a small amount of intra-peritoneal contrast due to retraction of the patient’s outside hospital placed external biliary drain.(B)Choledochoscopic image demonstrating one of many frond-like masses(arrow)within the intrahepatic bile ducts.The delivery wire of a Zero-Tip basket(Boston Scienti fic)is partially visualized.(C)Multiple additional areas of frond-like mass are seen within the bile ducts(arrows).An Amplatz Super Stiff wire(Boston Scienti fic)is partially seen.(D)The 5-French Bugbee fulgurating electrode(arrowhead)is seen coagulating a portion of the mass which is visibly charred(arrow).Portions of the mass are untreated on this image.(E)Additional fulguration with the electrode demonstrates complete charring of this mass prior to removal with a stone basket.(F)A Zero-Tip basket is used to remove portions of mass and stone(arrow)within the bile duct.

IPNB has a better prognosis than invasive cholangiocarcinoma.The prognosis worsens with increasing histologic grade of dysplasia.Patients with no histologic evidence of invasive carcinoma have a 5-year survival nearing 100%,while those with microinvasive or macroinvasive carcinoma have a 5-year survival of 17%and 14%,respectively[5].Even with surgical resection in the setting of invasive carcinoma,5-year survival has been reported as 38%[6].Although the sample obtained for histologic grading in our patient demonstrated high-grade IPNB,the presence of distant pulmonary metastases suggests a component of invasive carcinoma which was not sampled.In the literature,the median survival of patients with malignant IPNB with and without lymph node metastases has been reported as 12.1±5.1 months(95%CI:2.0–22.0)and 39.0±6.7 months(95%CI:25.9–52.1),respectively[5].In our patient the presence of distant pulmonary metastases is similarly associated with a guarded prognosis.However,the patient remains alive 5 months following the procedure.

Grounding pads were placed on the patient and the fluid used for choledochoscopy was changed from normal saline(Baxter;Troy,MI,USA)to 1.5%glycine(Baxter)in order to avoid electrocution.A 5-French,58 cm flexible Bugbee fulgurating electrode(Gyrus ACMI;Southborough,MA,USA)was then inserted through the working channel of the endoscope and used to perform monopolar electrocautery of the intra-biliary tumor using both coagulation and cutting modes(Fig.3D).Charring of the tumor could be readily visualized(Fig.3E).Additional detached fragments were retrieved using the Zero-Tip basket(Figs.3F and 4B).These samples were sent for pathologic analysis.After the tumor was sufficiently debulked,the endoscope was removed and a 14-French internal/external biliary drain was placed.Repeat contrast injection demonstrated decompression of the biliary tree.The patient’s clinical symptoms improved,and the serum bilirubin reduced to 9.8 mg/dL at 1-month follow-up after an additional left sided biliary drain was placed.

None.

(2) 试件滞回曲线呈梭形状且较为饱满，位移延性系数平均值为3.29，节点具有较好的耗能能力和抗震性能；

Fig.4.(A)Zero-Tip basket capturing a large mobile mass from the biliary tree.(B)Representative sample in formalin demonstrating large fragments of tumor from the biliary tree.

Contributors

This report was approved by the Institutional Review Board of University of Michigan Health System.

Funding

In this case,the patient was not a surgical candidate due to presumed lung metastases.Intrabiliary radiofrequency ablation has been previously reported to improve survival in patients with intrabiliary neoplasms including cholangiocarcinoma[7].Such techniques may be complicated by biliary tract perforation[8].Monopolar electrocautery facilitated fulguration,debulking and removal of intrabiliary neoplasm may be another method to maintain biliary patency in patients with biliary intraepithelial neoplasia and IPNB.

Ethical approval

SRaviN proposed the study.SRaviN and CJFB performed research and wrote the first draft.SRaviN and CJFB collected and analyzed the data.All authors contributed to the design and interpretation of the study and to further drafts.SRaviN is the guarantor.

The patient was placed under general anesthesia and antibiotic prophylaxis was administered with weight-based ceftriaxone(P fizer;New York City,USA).Initial cholangiography demonstrated severe dilatation of the intrahepatic biliary tree with complete obstruction at the level of the biliary hilum(Fig.2).A 4-French Cobra Glidecath(Terumo Interventional Systems;Tokyo,Japan)and Glidewire(Terumo Interventional Systems)were used to traverse the obstruction and gain access to the duodenum.Exchange was made for an Amplatz Super Stiff wire(Boston Scienti fic;Marlborough,MA,USA)and a second safety wire was placed.The percutaneous tract was dilated and a 20-French Peel-Away Sheath(Cook Medical;Bloomington,IN,USA)was placed into the intrahepatic bile ducts.A 16.5-French flexible endoscope(Olympus Corporation;Center Valley,PA,USA)with 5.5-French working channel was advanced into the biliary tree for choledochoscopy(Fig.3A).A freely mobile frond-like mass was encountered(Fig.3B),which was removed using a 3-French Zero-Tip basket(Boston Scienti fic)(Fig.4A).The sample was sent for frozen section which returned as biliary intraepithelial neoplasia 3(BilIN3).Subsequent final pathology revealed IPNB with high grade dysplasia.Choledochoscopic evaluation demonstrated numerous bulky wall-adherent frond-like masses invading the intrahepatic bile ducts(Fig.3C).Attempts were made to obtain additional samples of wall-adherent areas using the Zero-Tip basket and 5.5-French biopsy forceps(Cordis Corporation;Milipitas,CA,USA).However,only miniscule fragments could be obtained.

Competing interest

No bene fits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article.

References

[1]Wan XS,Xu YY,Qian JY,Yang XB,Wang AQ,He L,et al.Intraductal papillary neoplasm of the bile duct.World J Gastroenterol 2013;19:8595–8604.

[2]Rocha FG,Lee H,Katabi N,DeMatteo RP,Fong Y,D’Angelica MI,et al.Intraductal papillary neoplasm of the bile duct:a biliary equivalent to intraductal papillary mucinous neoplasm of the pancreas?Hepatology 2012;56:1352–1360.

[3]Yeh TS,Tseng JH,Chen TC,Liu NJ,Chiu CT,Jan YY,et al.Characterization of intrahepatic cholangiocarcinoma of the intraductal growth-type and its precursor lesions.Hepatology 2005;42:657–664.

[4]Jung G,Park KM,Lee SS,Yu E,Hong SM,Kim J.Long-term clinical outcome of the surgically resected intraductal papillary neoplasm of the bile duct.J Hepatol 2012;57:787–793.

[5]Yeh TS,Tseng JH,Chiu CT,Liu NJ,Chen TC,Jan YY,et al.Cholangiographic spectrum of intraductal papillary mucinous neoplasm of the bile ducts.Ann Surg 2006;244:248–253.

[6]Barton JG,Barrett DA,Maricevich MA,Schnelldorfer T,Wood CM,Smyrk TC,et al.Intraductal papillary mucinous neoplasm of the biliary tract:a real disease?HPB(Oxford)2009;11:684–691.

[7]Alis H,Sengoz C,Gonenc M,Kalayci MU,Kocatas A.Endobiliary radiofrequency ablation for malignant biliary obstruction.Hepatobiliary Pancreat Dis Int 2013;12:423–427.

[8]Zhou C,Wei B,Gao K,Zhai R.Biliary tract perforation following percutaneous endobiliary radiofrequency ablation:a report of two cases.Oncol Lett 2016;11:3813–3816.