An 81-year-old woman was referred to our department due to severe cholangitis after
laparoscopic cholecystectomy. She had a high fever and epigastric pain. Blood tests
showed elevated levels of C-reactive protein, hepatobiliary enzymes, and bilirubin.
Computed tomography and magnetic resonance imaging showed edematous change around
major duodenal papilla and there was suspicion of small stones in the bile duct (BD)
([Fig. 1]). These findings suggested acute cholangitis; therefore, we performed endoscopic
biliary drainage. We found periampullary diverticulum and papillary orifice (PO) aligned
downward due to a remarkably enlarged papillary mound ([Fig. 2a], [Fig. 2b], [Video 1]). We failed to perform deep cannulation in either the BD or pancreatic duct. During
the wire-guided cannulation, we observed the excretion of white milky discharge ([Fig. 2c]). We performed needle knife sphincterotomy. Despite pre-cutting, deep cannulation
was not completed. Finally, we decided to perform a rendezvous method of percutaneous
transhepatic biliary drainage on the second day of hospitalization. We punctured the
intrahepatic BD and advanced the guidewire into the duodenum via the papilla. Surprisingly,
the guidewire stuck out of another concavity, which was different from what we thought
was the PO. Thus, the concavity where the guidewire stuck out revealed to be a “real
PO” ([Fig. 2d]). Despite sweeps by balloon catheter, there was no excretion of milky fluid. After
this procedure, the patient’s symptoms promptly improved and she had no adverse events.
Fig. 1 Abdominal computed tomography (CT) and magnetic resonance (MR) imaging of major duodenal
papilla and the bile duct (BD). a Edematous change was suspected around the major duodenal papilla on axial CT. b Thickening of the BD wall was seen on coronal CT. c T2-weighted MR image also suggested edematous changes in the major duodenal papilla.
d MR cholangiography showed something like sludge (arrow) in the BD.
Fig. 2 a Papilla viewed from above. The periampullary diverticulum and enlarged papillary
mound were seen. b Papilla viewed from below. What we initially thought was the papillary orifice (PO)
(*) was seen under the covering fold (X). c Milky fluid excretion from the PO during cannulation. d The real PO (arrow) were identified after a percutaneous rendezvous technique. Whitish
ulcerative mucosa indicates the site that we negotiated and which precut revealed
to be a “pseudo PO.” e Real PO (arrow) and pseudo-PO (*) 6 months after the procedures.
Video 1 74-year-old woman with cholangitis in whom we failed to perform endoscopic biliary
drainage despite wire- and contrast-guided cannulation and needle knife sphincterotomy.
Rendezvous cannulation using percutaneous transhepatic biliary drainage was performed.
What was the “pseudo-PO”? In this case, we could not histologically diagnose that
lesion. However, the excretion of milky fluid was reported in a case of lymphangioma
[1]
[2], but these are rare. A Brunner’s gland hyperplasia [3], mucus secreting polyp [4], and ectopic pancreatic tissue [5] have been known to sometimes have an orifice structure. The excretion of milky fluid
by guidewire penetration may indicate lymphatic retention near the papilla. We considered
that submucosal lymphatics compressed the distal segment of BD, which could cause
cholangitis.
