Recurrent Cholangitis by Biliary Stasis Due to Non-Obstructive Afferent Loop Syndrome After Pylorus-Preserving Pancreatoduodenectomy: Report of a Case

We report a 71-year-old man who had undergone pylorus-preserving pancreatoduodenectomy (PPPD) using PPPD-IV reconstruction for cholangiocarcinoma. For 6 years thereafter, he had suffered recurrent cholangitis, and also a right liver abscess (S5/8), which required percutaneous drainage at 9 years after PPPD. At 16 years after PPPD, he had been admitted to the other hospital because of acute purulent cholangitis. Although medical treatment resolved the cholangitis, the patient was referred to our hospital because of dilatation of the intrahepatic biliary duct (B2). Peroral double-balloon enteroscopy revealed that the diameter of the hepaticojejunostomy anastomosis was 12 mm, and cholangiography detected intrahepatic stones. Lithotripsy was performed using a basket catheter. At 1 year after lithotripsy procedure, the patient is doing well. Hepatobiliary scintigraphy at 60 minutes after intravenous injection demonstrated that deposit of the tracer still remained in the upper afferent loop jejunum. Therefore, we considered that the recurrent cholangitis, liver abscess, and intrahepatic lithiasis have been caused by biliary stasis due to nonobstructive afferent loop syndrome. Biliary retention due to nonobstructive afferent loop syndrome may cause recurrent cholangitis or liver abscess after hepaticojejunostomy, and double-balloon enteroscopy and hepatobiliary scintigraphy are useful for the diagnosis of nonobstructive afferent loop syndrome.Key words: Nonobstructive afferent loop syndrome, Biliary stasis, Hepaticojejunostomy, Hepatobiliary scintigraphy, Double-balloon enteroscopyIt has been reported that cholangitis occurs in between 6.7% and 14.3% of postoperative pancreatoduodenectomy (PD).¹ Most cases of cholangitis originate due to biliary stasis, which is broadly caused by either anastomotic or nonanastomotic stenosis. In many cases, anastomotic stenosis is accompanied by intrahepatic biliary duct dilatation and obstructive jaundice, making early diagnosis and treatment possible.^2–3 On the other hand, nonanastomotic stenosis, including those of afferent loop syndrome, is performed as a conservative treatment for unexplained fever and cholangitis. However, in many cases, the cause remains unidentified, thereby causing this condition to repeat itself. Since cholangitis can at times be fatal, it is therefore important to identify the cause.It has been reported that afferent loop syndrome occurs in around 13% of postoperative PD patients.⁴ Afferent loop syndrome is generally caused by mechanical occlusion due to the recurrence or metastasis of cancer,^4–6 adhesion,^7–8 torsion,⁹ internal hernia,¹⁰ enterolithiasis,^11–12 etc., and thereafter, leads to a syndrome associated with acute abdominal symptom or acute cholangitis. On the other hand, nonobstructive afferent loop syndrome may also be caused by biliary stasis due to jejunal motility failure or the length of the blind end or jejunum, and thereafter, leads to acute cholangitis, liver abscess, and the formation of enterolithiasis and intrahepatic stones. Nonobstructive afferent loop syndrome occurs in around 37% of all of the afferent loop syndrome,^12–13 but few cases have actually been reported.We herein report a rare case in which the patient experienced recurrent cholangitis and liver abscess by biliary stasis due to nonobstructive afferent loop syndrome after pylorus-preserving pancreatoduodenectomy (PPPD) for cholangiocarcinoma.