Bouveret's syndrome is a rare condition of gastric outlet obstruction resulting from the migration of a gallstone through a choledochoduodenal fistula. Due to the large size of these stones and the difficult location in which they become impacted, endoscopic treatment is unsuccessful and most patients require surgery. We report the case of an elderly male who presented with nausea and hematemesis, and was found on CT scan and endoscopy to have an obstructing gallstone in his duodenal bulb. After several endoscopic sessions and the use of multiple instruments including a Holmium: YAG laser and electrohydraulic lithotripter, fragmentation and endoscopic removal of the stone were successful. We believe this to be the first case of Bouveret's syndrome successfully treated by endoscopy alone in the United States. We describe the difficulties encountered which necessitated varied and innovative therapeutic techniques.
Gastric outlet obstruction by a
gallstone migrating through a cholecystoduodenal
fistula (Bouveret’s syndrome) is a very rare condition, accounting for fewer
than 5% of cases of gallstone ileus [
An 85-year-old male with advanced
Alzheimer’s dementia, diabetes mellitus, and atrial fibrillation presented with
several days of nausea, vomiting, and lethargy. There was no report of
abdominal pain, fever, or chills. His
vital signs were stable and his abdominal exam benign. Nasogastric lavage was significant for one liter
of coffee-ground material. His laboratory
examination demonstrated a white blood cell
count of 25 000/cm2, Hematocrit of 33%, creatinine of 1.5 mg/dL, and normal liver enzymes. A CT scan (see
Figure
Computed tomography (CT) scan of abdomen and pelvis. (a) Axial image showing pneumobilia (arrow) and a dilated fluid-filled stomach (
Endoscopy demonstrated old blood in
a distended stomach, and a large gallstone in the duodenal bulb obstructing the
pylorus (see Figure
(a) large gallstone in the duodenal bulb, obstructing the pylorus. (b) Attempts to extract the stone failed with multiple instruments, including biliary and CRE balloons (
Two different lithotripters were used to fragment the stone. (a) Holmium: YAG laser produced small cracks on the proximal surface (arrows) but the majority of the stone still remained impacted. (b) Electrohydraulic lithotripsy (IEHL) successfully shattered the outer “shell” of the stone (white arrow) and left behind a smaller, much harder core (black arrow). (c) Ultimately, the majority of the stone was fragmented after extensive use of both lithotriptors.
Three days later endoscopy was
repeated, and an intracorporeal electrohydraulic lithotripter (IEHL; Northgate
Technologies, Ill, USA), which was previously unavailable, was employed, as
working with the Holmium: YAG laser had been only partially successful. Using a 1.9F fiber (power of 1, increased to
40; frequency of 10, increased to 30) under constant saline irrigation, IEHL
was successful at shattering the outer “shell” of the stone and breaking it
into two large pieces, leaving behind an extremely hard, smaller core (see Figure
Double-snare extraction technique. Two overlapping jumbo polypectomy snares (arrows) were used to grasp the stone at different angles, providing adequate leverage for extraction into the stomach.
Visualization of the choledochoduodenal fistula. (a) After stone extraction, the large orifice of the fistula (arrow) can be seen in the duodenal bulb, whose mucosa is diffusely ulcerated. (b) The gastroscope passed easily through the fistula into the lumen of the gallbladder.
On repeat endoscopy, the largest
stone fragment had again become impacted in the duodenal bulb, but was
extracted by placing a biliary balloon behind it and a polypectomy snare around
its center. In the stomach, the largest
stones could not be crushed despite use of a mechanical lithotripter. To
further break up the stones, we used the Holmium: YAG laser (1000-micron fiber for 128 joules for 427 pulses
per second for a total delivery time of 2 minutes and 49 seconds) to bore multiple
holes into the center of each fragment, which was then crushed with a biliary stone
basket. The larger fragments were
removed perorally (see Figure
The largest stone fragments were removed perorally. The inner composition of the largest piece can be seen, measuring greater than 1 cm in diameter.
Bouveret’s syndrome was first
described in 1896 by Leon Bouveret, a French internist and masterful
diagnostician, who reported two patients with large gallstones causing gastric
outlet obstruction, both of whom died [
Our case is representative of the typical patient with Bouveret’s, as well as the difficult challenges involved in approaching the endoscopic management. Due to multiple comorbidities and a severely ulcerated duodenum, the surgeons were hesitant to operate. The large size of the stone (more than 3 cm in diameter), its location, and the propensity for instruments to follow the fistulous tract rather than the duodenal lumen made maneuvering extremely difficult, and precluded the use of a mechanical lithotripter.
We chose the Holmium: YAG laser and IEHL because of their prior successes in treating difficult common bile duct stones, as well as their availability at our institution. We found the Holmium: YAG laser to be useful in producing initial cracks in the stone’s surface; IEHL, however, seemed to be more effective in uniformly shattering the outer surface though not very effective at attacking its harder, inner core. Using both lithotripters required a significant amount of time, in part because of the minimal working space as well as the extreme care involved in avoiding further damage to the already ulcerated bulbar mucosa. Using laser or IEHL fibers of larger diameter may have improved the efficiency of lithotripsy, but these were not available to us. When approaching the large stone fragments in the stomach, we found “drilling” multiple holes with the Holmium: YAG laser to weaken the internal structure of the stone’s core to be a very useful technique which then allowed us to easily crush these large fragments with a basket. We also found that to successfully extract the stone from the bulb after lithotripsy, a single instrument did not provide enough leverage or balance. Using two instruments with a double-channel gastroscope, however, was proved to be successful twice (once using two snares, once using a snare and a biliary balloon).
Another important observation in
our case was that between the second and third endoscopies, one of the stone fragments
left in the stomach again became impacted in the duodenal bulb. Fortunately, this happened to be quite a
large fragment, for a smaller piece might have traversed the duodenum and
lodged in the ileum causing a distal gallstone ileus requiring surgery. This complication has indeed been reported several
times in the literature [
We believe our case to be the first full-length report of the successful treatment in the United States of Bouveret’s syndrome with endoscopy alone, as well as a unique description of the complementary use of different lithotriptors and instruments. Our experience suggests that endoscopists faced with this clinical problem should use a double channel gastroscope, initiate stone fragmentation with IEHL as first line management, crush stone fragments that are left in the stomach to prevent ileal obstruction, and expect multiple endoscopic sessions. We anticipate that due to the aging population in the US and the epidemic of obesity, Bouveret’s will likely become more common than previously reported. It is, therefore, important for endoscopists to be familiar with the multiple options available to effectively treat these difficult cases without surgery.