Recently, biliary tract intraductal papillary mucinous neoplasm (IPMN) was described
as a rare but important preinvasive lesion coinciding with pancreatic IPMN [1]. Several studies have revealed dysregulated molecular pathways involving mutations
of the GNAS and KRAS genes and have tried to distinguish between biliary IPMN and pancreatic IPMN by means
of their mucin-subtype pattern [2]. Nevertheless, the majority of data on biliary tract IPMNs come from retrospective
studies, mostly where surgical interventions were performed [3]. Only one case report has so far described a combined chemoradiotherapy approach
in a patient with synchronous biliary tract and pancreatic IPMNs [4].
Here we present the complex case of a patient with a biliary tract IPMN that, after
initial surgery, was successfully treated by intraductal transhepatic radiofrequency
ablation (RFA) via a transcutaneous access route ([Video 1]). The 71-year-old man had a diagnosis of arterial hypertension and type 2 diabetes
mellitus. The patient was transferred from a primary care hospital. Initially, he
underwent a laparoscopic cholecystectomy because of cholecystitis. A revision of the
bile duct with T-drain implantation and subhepatic drains was necessary because of
postsurgical bile duct leakage. A computed tomography (CT) scan was suspicious for
an intrahepatic tumor but was not reproducible. Initially, we visualized the bile
duct via the T-drain and found contrast defects that were felt likely to be bile duct
stones. However, endoscopic retrograde cholangiopancreatography (ERCP) found no calculi,
although viscous mucus was removed by means of a balloon and a basket catheter. Interestingly,
magnetic resonance cholangiopancreatography (MRCP) revealed an inconspicuous bile
duct.
Video 1 The course and procedures performed for a patient with biliary duct intraductal papillary
mucinous neoplasm (IPMN). After undergoing an initial hemihepatectomy, the patient
suffered from recurrent mucin production and cholangitis. Transhepatic cholangioscopy
showed a biliary IPMN in the remaining bile duct, which was treated by intraductal
radiofrequency ablation (RFA).
In the following weeks, the patient suffered from relapsing cholangitis due to episodes
of mucus-induced bile duct stent occlusion. Mother-baby cholangioscopy was infeasible
because the viscous mucus could not be removed. Finally, ERCP showed a stenosis of
the right intrahepatic bile duct that revealed signal enhancement on a positron emission
tomography-CT (PET-CT) scan.
Following an interdisciplinary meeting, we recommended to the patient that he undergo
a right-sided hemihepatectomy for a tentative diagnosis of a mucin-producing tumor,
and this was subsequently performed without difficulty. However, the histology showed
a low grade biliary IPMN with an R1 resection.
In the next few months, the patient suffered from recurrent bile duct stent occlusions,
cholangitis, and bilioma. We therefore decided, owing to unsatisfactory ERCP treatment,
to perform left-sided percutaneous transhepatic cholangiodrainage (PTCD). Over a period
of 6 weeks, this transhepatic access to the bile duct was dilated to 18-Fr. Thus,
we created a large enough approach tract to be able to insert a small diameter endoscope
and conduct a wire-guided transhepatic cholangioscopy. Through this, we were able
to observe and confirm the presence of a biliary IPMN in the remaining bile duct.
In addition, transhepatic intraductal ablation of the biliary IPMN was successfully
performed by RFA using the Habib EndoHPB ablation catheter, with no immediate or delayed
complications. Following the RFA treatment, the patient has shown no signs of cholangitis
or recurrent biliary IPMN during 1 year of follow-up.
To the best of our knowledge, this is the first report of transhepatic intraductal
RFA of a biliary duct IPMN performed via transcutaneous access. However, randomized
controlled trials will be necessary to further assess this interventional technique.
Endoscopy_UCTN_Code_TTT_1AR_2AF
Endoscopy E-Videos is a free access online section, reporting on interesting cases and new techniques
in gastroenterological endoscopy. All papers include a high
quality video and all contributions are
freely accessible online.
This section has its own submission
website at
https://mc.manuscriptcentral.com/e-videos
