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Case Report Clinics in Surgery Published: 16 Mar, 2021

Hybrid Surgical and Endoscopic Transjejunal ERCP approach for Intra-Hepatic Biliary Stones Secondary to Recurrent Roux-En-Y Hepaticojejunal Anastomotic Strictures

Nolasco Vaz J1*, Faria A1, Gaio R2, Maria B1, Felício C1, Noronha Ferreira C3, Carrilho-Ribeiro L3, Tato Marinho R3 and Coutinho J1,3 1Department of General Surgery, Centro Hospitalar Universitário Lisboa Norte, Portugal

2Department of , Centro Hospitalar Universitário Lisboa Norte, Portugal

3Department of Gastroenterology, Centro Hospitalar Universitário Lisboa Norte, Portugal

Abstract Hepaticojejunal anastomotic stricture can result in intra-hepatic biliary stones and recurrent cholangitis. In the presence of altered small bowel anatomy, therapeutic ERCP has a low success rate. In patients with gastric bypass, there is increased expertise and efficiency in performing ERCP through transgastric access. Transjejunal access is an alternative in the absence of a gastric remnant. We present a case of a 30-years-old female with recurrent cholangitis and intra-hepatic biliary stones resulting from hepaticojejunal anastomotic stricture. The presence of severe adhesions between jejunal loops resulted in failure of double balloon assisted ERCP. A hybrid surgical- endoscopic approach via transjejunal enterotomy allowed access to hepaticojejunal anastomotic stricture which was dilated with through the scope balloon and subsequent removal of multiple biliary stones. OPEN ACCESS A hybrid surgical-endoscopic transjejunal ERCP is a feasible approach after failure of other *Correspondence: conventional endoscopic approaches to manage patients with hepaticojejunal anastomotic strictures. Nolasco Vaz J, Department of Keywords: Transjejunal ERCP; Roux-en-Y; Hepaticojejunal anastomotic strictures; Intra- General Surgery, Centro Hospitalar hepatic biliary stones Universitário Lisboa Norte, Portugal, Tel: 00351963157940; Abbreviations E-mail: [email protected] Received Date: 01 Feb 2021 ERCP: Endoscopic retrograde cholangiopancreatography; DBE: Double balloon enteroscopy; DBE-ERCP: Double-Balloon Enteroscopy-assisted Endoscopic Retrograde Accepted Date: 05 Mar 2021 Cholangiopancreatography; MRCP: Magnetic Resonance Cholangiopancreatography; IHBD: Intra- Published Date: 16 Mar 2021 Hepatic ; TTS: Through the Scope; PTC: Percutaneous Transhepatic ; Citation: RYHJ: Roux-en-Y Hepaticojejunal Nolasco Vaz J, Faria A, Gaio R, Maria Introduction B, Felício C, Noronha Ferreira C, et al. Hybrid Surgical and Endoscopic Strictures of bilioenteric anastomosis occur in 2.6% to 30% of patients [1–4]. The mean time Transjejunal ERCP approach for Intra- for stricture development varies between 13 and 21.6 months, being more frequent in proximal Hepatic Biliary Stones Secondary to reconstructions, after iatrogenic injury and in younger patients [1,3,4]. Bilioenteric anastomotic Recurrent Roux-En-Y Hepaticojejunal strictures have been described in up to 85% of all patients with intrahepatic lithiasis, being more Anastomotic Strictures. Clin Surg. frequent in the left biliary duct and result in recurrent cholangitis, and possible atrophy and 2021; 6: 3109. cholangiocarcinoma [5–7]. Copyright © 2021 Nolasco Vaz J. This Conventional ERCP has success rates of 90% to 95% in the management of biliary duct lithiasis is an open access article distributed [8]. In patients with altered bowel anatomy with Roux-en-Y reconstruction, the therapeutic success under the Creative Commons rate of Double Balloon Enteroscopy (DBE) assisted ERCP decreases to around 70% [9]. Attribution License, which permits We describe the use of a hybrid surgical and endoscopic technique via transjejunal approach unrestricted use, distribution, and to manage hepaticojejunal anastomotic stricture and intrahepatic biliary stones in a patient with a reproduction in any medium, provided Roux-en-Y reconstruction. This hybrid technique is a feasible alternative when DBE assisted ERCP the original work is properly cited. fails.

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Case Presentation We present the case of a 30-year-old female patient from Guinea- Bissau, who underwent at 2 years of age, surgical excision of a hepatic hamartoma (25 cm × 20 cm), complicated with left hepatic duct , managed with a Roux-en-Y Hepaticojejunal (RYHJ) anastomosis of the right and left hepatic ducts. The patient developed recurrent cholangitis from the age of 13 and was transferred to Portugal for medical management at 28 years of age. Evaluation with Magnetic Resonance Cholangiopancreatography (MRCP) revealed indefinite anastomosis, Intra-Hepatic Bile Duct (IHBD) dilation and several intraluminal filling defects in both left and right lobes (Figure 1). Figure 3: First follow-up MRI after initial DBE-ERCP. Axial FSE T2-WI- FS images (upper row) and corresponding MRCP images (lower row) She was hospitalized with obstructive jaundice (total bilirubin14 demonstrate only mild reduction of intra-hepatic biliary duct dilation in the left mg/dL) and underwent urgent DBE-ERCP. The hepaticojejunal lobe (orange arrows in a and c) and persistent significant dilation and multiple anastomosis was identified and strictures of both right and left filling defects (arrows) in left IHBD (aanda.1), at the confluence of left and right bile ducts (bandb.1) and in right IHBD (c and c.1). hepaticojejunal anastomosis with anastomotic orifices measuring 4 and 2 mm were observed. Through the scope balloon dilation up to 9 mm of both strictures was performed with incomplete removal of left hepatic duct calculi (2 residual calculi in the proximal left hepatic duct, inaccessible to the stone removal balloon catheter) (Figure 2).

Figure 4: Left and right hepaticojejunostomy.

The patient maintained cholestatic jaundice and a new MRCP confirmed persistence of IHBD dilation and IHBD lithiasis in both left and right lobes (Figure 3). A second DBE-ERCP was performed 4 months later and a recurrent stricture of the right and left hepaticojejunal anastomosis measuring 5 mm and 2 mm were observed (Figure 4), which were again dilatated 4 with TTS balloon till 10 mm and 8 mm, respectively. At the end of the second ERCP, there were no apparent intra-hepatic biliary repletion defects on cholangiography. After the procedure there was improvement of total bilirubin which however persisted above 6 mg/dL. The patient was lost to follow-up, but 16 months later, was Figure 1: Initial MRI. MRCP (a) and axial FSE T2-WI-FS images (b, c and d) hospitalized with acute cholangitis with a total bilirubin of 10.2 mg/ demonstrate dilation and multiple filling defects (arrows) in left intra-hepatic biliary ducts (orange), main left hepatic duct and confluence (white) and right dL. An urgent DBE assisted ERCP was performed but access to the posterior biliary duct (blue). bilioenteric anastomosis was this time not possible probably due to small bowel adhesions. During the procedure it was possible to identify the biliopancreatic limb and this was tattooed with India ink to facilitated identification for subsequent procedures. After evaluation in a multidisciplinary meeting, it was decided to perform a hybrid open surgical-endoscopic technique to manage the patient. The presence of multiple adhesions in the final 10 cm ofthe hepatobiliary limb and a total length longer than 150 cm precluded manual guidance of a colonoscope introduced orally. Therefore we opted for an enterotomy with stay sutures, 10 cm away from the hepaticojejunal anastomosis to enable endoscopic access (Figures 5-7). The maintenance of a sterile field was warranted with surgical drapes around the enterotomy, a laparoscopic camera drape around Figure 2: Right hepaticojejunostomy. the endoscope and the gastroenterologist wearing sterile gloves and

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Figure 8: Guide wire inside right hepatic duct.

Figure 5: Enterotomy with stay suture.

Figure 9: Pigment lithiasis.

Figure 6: Endoscopic access within sterile laparoscopic camera drape.

Figure 10: Follow-up MRI after hybrid surgical-endoscopic technique. Axial FSE T2-WI images (upper row) and MRCP images (lower row) demonstrate persistent intra-hepatic biliary duct dilation and filling defects (orange arrows in a, b and d) in the left lobe but significant reduction of right IHBD dilation Figure 7: Operating theater distribution. (blue arrow in c), even though some filling defects can be seen in the right intra-hepatic biliary tree. Note there is also resolution of the filling defects in gown. the right bile duct near the confluence (green arrow in E).

This technique allowed diagnostic and therapeutic approach of anastomotic stricture (Figure 11) and allowed dilation with a TTS a right duct hepatic stenosis (5 mm) with balloon dilation (up to balloon until 10 mm and multiple calculi removal from left IHBD 11 mm) and calculi removal from right IHBD (Figure 8,9). The left (Figure 12,13). A cholangioscopy with a pediatric endoscope IHBD had no apparent filling defect on cholangiography. excluded the presence of residual lithiasis in IHBD. An intra- Due to persistence of jaundice a new MRCP was performed procedural ruled out secondary biliary cirrhosis due to which showed evidence of left IHBD stones (Figure 10). The patient recurrent hepaticojejunal anastomotic strictures. was re-operated 3 weeks later, repeating the hybrid approach. The The patient was discharged clinically well but due to persistent presence of the India ink tattoo enabled an easier identification of total serum bilirubin values of 3 mg/dL, MRCP was repeated and the biliopancreatic limb in the presence of firm and dense adhesions. it showed 3 peripheral left IHBD calculi with left IHBD persistent Transjejunal endoscopic ERCP confirmed tight left hepaticojejunal dilation.

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Surgical treatment of bilioenteric anastomotic stricture and secondary intra-hepatic biliary stones is complex; requiring expertise from high volume centers and despite this is often associated with considerable morbidity including intra-abdominal collections, biliary/anastomotic and recurrent strictures [13-18]. In patients with RYHJ stricture and recurrent intrahepatic lithiasis, sub parietal or subcutaneous hepaticojejunal loop allows permanent and easier access to biliary instrumentation with minimal morbidity [6,7]. ERCP is one of the most effective diagnostic and therapeutic modalities in patients with biliary disease [19]. In the presence of surgically altered gastrointestinal anatomy with a long Roux-en-Y loop, the use of pediatric or adult colonoscopes or enteroscopic assisted ERCP is recommended [19-24]. In patients with altered Figure 11: Left HJ stricture. anatomy ERCP is challenging due to 1) difficulty in identification of the biliopancreatic limb 2) challenges in identification and cannulation of the papilla/bilioenteric anastomosis from an altered position 3) therapeutic limitations imposed by the lack of side view, elevator and compatible devices usually designed for standard ERCP [21-24]. Identifying the biliopancreatic limb can be complex, especially in the presence of adhesions but, as in the present report, India ink tattoo can help overcome this difficulty [25,26]. Saleem et al. [26] used transoral endoscopic insertion to identify the afferent limb. Long total limb length and sharp angulations secondary to adherences can preclude ERCP success and increase the risk of complications [19,22,24,27]. Shorter Roux-en-Y reconstructions increase the success rates of access to the papilla/bilioenteric anastomosis. The therapeutic success rate of enteroscopy assisted ERCP drops from 88% to 33% Figure 12: Bilateral calculi removal. with total Roux + biliopancreatic limb length under 150 cm or between 150 cm to 250 cm, respectively [28]. Percutaneous transhepatic biliary drainage alone or with endoscopic rendezvous is an alternative and useful technique for emergent biliary decompression in patients with IHBD dilation [19,24,27]. Schapira et al. [29], first described in 1975 an and retrograde cholangiography via gastrostomy, resulting in a shorter route to the papilla/bilioenteric anastomosis and overcoming proximal anatomical obstacles [30-32]. Combined surgical and endoscopic approach either open or laparoscopic, enable therapeutic success in the presence of complex altered anatomy [21]. The increase in the incidence of morbid obesity and has resulted in more frequent transgastric ERCP procedures which are also cost-effective in patients with long Roux + hepatobiliary limb length [21,28,33,34]. A recent multicentric study with 579 Figure 13: Patent left and right HJ anastomosis. -assisted ERCP after gastric bypass, revealed success rates comparable with standard ERCP in normal anatomy patients, with Their peripheral intrahepatic location precluded cholangioscopic total procedure time of 152 min [33]. approach and left has been planned if she develops EUS-directed transgastric or transenteric ERCP with lumen- recurrent cholangitis. apposing metal stent is gaining popularity [35], but it remains Discussion technically challenging with potentially serious adverse events, such as stent migration and perforation and can be associated with weight Endoscopic treatment is currently the gold standard for regain in RYGB for morbid obesity [36,37]. management of benign biliary strictures approach, with percutaneous or surgical interventions being indicated for those patients with failed Surdeanu et al. [38] report a case where dense adhesions endoscopic procedures [10-12]. precluded gastric remnant gastrostomy construction, resulting in the

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Table 1: Transjejunal ERCP case reports in the literature. Transjejunal ERCP Author Sex Age GI anatomy Indication for ERCP Adhesions Morbidities Result approach Asbun [11] F 36 RYHJ HJ stricture → cholangitis Open Yes 0 R

M 45 RYHJ HJ stricture→cholangitis Open Yes 0 R

F 33 RYHJ to segment III Intra-hepaticlithiasis→cholangitis Lap→Open Yes 0 R RYGB→ Recurrent pancreatitis + papillary F 34 Transduodenal Open - 0 R Mergener stricture sphincteroplasty 44 F 35 DS Minorpapillastricture→pancreatitis Open - - R D.3 Mutignani 45 F 63 BPD Choledocholithiasis Lap (via trocar) - 0 days Sphincter of Oddi dysfunction→ Pneumothorax Liver abscess Lopes 46 F 18 RY partial Lap (via trocar) No R cholestasis Right PV thrombosis RY subtotal Lap (mini- D.5 Saleem [26] F 45 Pancreaticopleural fistula Yes 0 gastrectomy ) days HJ stricture →intra-hepatic lithiasis Lap (mini- D.5 Mansor [25] F 17 RYHJ Yes 0 → cholangitis laparotomy) days Surdeanu Lap (mini- D.7 M 50 RYGB Choledocholithiasis Yes 0 [38] laparotomy) days DS → reversal with M 60 Choledocholithiasis Lap(via trocar) - - R Marchesini enteroanastomosis 47 F 52 DS Choledocholithiasis Lap(via trocar) - - R RY subtotal Lap (mini- D.6 Mita 48 F 79 Choledocholithiasis Yes 0 gastrectomy laparotomy) days D. 4 Dalmonte 41 F Young RYGB Choledocholithiasis Lap(mini-laparotomy) - 0 days Baca-Arzaga D.3 F 50 RYHJ HJ stricture→ Intra-hepatic lithiasis Lap(via trocar) - 0 46 days F: Female; M: Male; GI: Gastrointestinal; RYHJ: Roux-en-Y Hepaticojejunostomy; RYGB: Roux-en-Y Gastric Bypass; DS: ; BPD RY: Roux-en-Y; HJ: Hepaticojejunostomy; ERCP: Endoscopic Retrograde Cholangiopancreatography; Lap: Laparoscopic; PV: Portal Vein; R: Recovered; D: Discharged need for a transjejunal access for choledocholithiasis resolution with ERCP, and most were discharged after 3 to 7 days. There is only one laparoscopic assisted ERCP. case mentioning associated morbidity. Lopes et al. [39] describe a patient with Roux-en-Y partial Conclusion gastrectomy with symptomatic cholestasis secondary to sphincter of In conclusion, we believe that a hybrid surgical-endoscopic Oddi dysfunction, with a failed approach either with conventional transjejunal ERCP is a safe technique that can be useful after failure ERCP either with percutaneous transhepatic endoscopic rendezvous. of conventional endoscopic techniques including DBE assisted ERCP In this case, a hybrid endoscopic laparoscopy assisted technique via in managing patients with RYHJ strictures. biliopancreatic limb enterotomy allowed the access to the papilla using a 15 mm trocar. References Transjejunal ERCP allows overtaking anatomical barriers like 1. House MG, Cameron JL, Schulick RD, Campbell KA, Sauter PK, long limb distances or tortuous bowel secondary to adhesions. It Coleman J, et al. 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