Keywords
intradiverticular papilla - Lemmel's syndrome - duodenal diverticulum
Introduction
The periampullary diverticulum (PAD) is a duodenal outpouching near the major duodenal
papilla. PAD is seen in 9% to 32.8% of patients undergoing endoscopic retrograde cholangiopancreatography
(ERCP).[1] The majority of the PAD are asymptomatic and are diagnosed incidentally during computed
tomography (CT) or ERCP. In rare instances, they can cause pancreaticobiliary complications
such as obstructive jaundice because of the mechanical effect of the diverticulum
(Lemmel's syndrome).[2] Apart from the clinical significance, they often pose technical challenges during
the ERCP. The papillary opening is often seen either at the superior or the inferior
border of the PAD. However, when the papilla is entirely within the PAD, the orientation
changes making cannulation of the bile duct problematic.
In this case report, we look at a novel way to get around this problem by using a
band ligator to uncover the papilla out of PAD.
Case Report
A 40-year-old man presented with features of obstructive jaundice, abdominal pain,
and fever for the past 2 weeks. His physical examination revealed icterus and mild
tenderness in the right hypochondrium. Laboratory investigations showed elevated total
counts (15,000 cells/mm3) total bilirubin of 9.8 mg/dL, direct bilirubin 6.38 mg/dL, SGOT 83 IU/L, SGPT 103 IU/L,
alkaline phosphate 481 IU/L, and gamma GT 566 U/L.
A CT scan of the abdomen showed a periampullary diverticulum measuring 2 cm × 1 cm
in the second part of the duodenum compressing the intrapancreatic distal common bile
duct (CBD) causing upstream dilatation of the CBD, common hepatic duct (CHD), and
intrahepatic biliary radicles (IHBR). No calculi were seen in the biliary system.
The above features were suggestive of Lemmel's syndrome.
The patient was started on broad-spectrum antibiotic coverage along with intravenous
fluids. He was taken up for an emergency ERCP to relieve the biliary obstruction.
During the procedure, two large diverticula were visualized on the medial wall of
the second part of the duodenum.
The ampullary ridge was identified at the mouth of one of the diverticulum but the
papilla could not be visualized. A biopsy forceps were used to pull and confirm the
position of the papillary opening within the diverticulum ([Fig. 1]). Biliary cannulation was attempted but was unsuccessful. A marking was made using
the same forceps at the ampullary ridge for later identification. The placement of
a hemoclip at the marked site was attempted but failed due to technical difficulties.
The duodenoscope was withdrawn entirely, and a forward view scope was introduced with
an EVL band ligator (COOK 6 Shooter® Universal Saeed® Multi-Band Ligator).
Fig. 1 (A) Periampullary diverticulum with the hidden intradiverticular papilla. (B) Papillary opening (black arrow) seen after pulling the caudal portion of diverticulum
below the ampullary ridge.
Two bands were deployed at the previously marked site ([Fig. 2A]) with the intent to evert the edge of the diverticulum within which the ampullary
opening was hidden. The duodenoscope was reintroduced, and the papillary opening was
clearly visible. The papilla was easily cannulated, and biliary access was obtained
([Fig. 2B]). Cholangiogram showed a distal CBD narrowing with dilatation of proximal CBD, CHD,
and IHBR, without any evidence of choledocholithiasis or stricture, thus confirming
Lemmel's syndrome ([Fig. 3]). A sphincterotomy was done and a CBD stent was placed.
Fig. 2 (A) Side view endoscopy with a papillary opening (white arrow) facing the duodenal lumen
after successfully applying two EVL bands (black arrow). (B). Successful cannulation of the papillary opening post EVL band application.
Fig. 3 Cholangiogram showing a distal CBD narrowing (white arrow) with dilatation of the
proximal CBD, CHD, and IHBR.
There were no complications either during or after the procedure. The patient's clinical
and biochemical status improved after the procedure, and he was discharged 3 days
later.
Discussion
The duodenal diverticulum is a mucosal or submucosal outpouching of the duodenal wall
with a partially weak muscle. They are most commonly found in the second part of the
duodenum near the periampullary region.[3] PAD is uncommon in young and increases in prevalence with age; the overall prevalence
among the elderly is estimated to be 65%.[4]
PAD is usually asymptomatic and discovered incidentally, but it can cause various
pathological manifestations, including CBD obstruction, pancreatitis, perforation,
bleeding, and, in rare cases, carcinoma.[5]
[6] Lemmel's syndrome was first described in 1934 as obstructive jaundice caused by
a periampullary duodenal diverticulum in the absence of choledocholithiasis. PAD causes
mechanical compression of the intrapancreatic CBD with resultant upstream dilatation
of the extra- and intrahepatic bile ducts.[2]
This syndrome presents a unique challenge for the endoscopist. The ampulla can be
difficult to visualize because it is buried within the diverticular wall. Identifying
and cannulating the ampulla can be done in a variety of ways, including two devices
in one-channel method, reversed guidewire method, double endoscope method, balloon
dilation of the narrow diverticular neck, endoclip-assisted cannulation, and cap-assisted
cannulation. The success rate of these methods ranges from 80% to 100% with complication
rates ranging from 4% to 25%.[7] However, these methods require either an additional accessory or technical expertise.
In our case, we used a simple and easily available EVL band ligator to retract the
ampulla from the PAD. After the band placement ampullary opening is easily visualized
and we achieved quick and easy cannulation in the first attempt. To the best of our
knowledge, this is the first time in the literature to use the EVL band ligator in
assisting ERCP. There are certain limitations with this method, exposure of ampulla
after the banding can only be assessed after we reintroduce the duodenoscope. Inadvertent
capturing of the full thickness of the diverticular wall, the risk of bleeding after
tissue necrosis, and the need for repeated banding during follow-up procedures should
also be kept in mind when attempting this technique. Further studies of this method
are needed to assess the clinical outcomes and complications.
In conclusion, the technique provides an alternative and elegant method of gaining
biliary access without the need to learn any additional skills.