Laparoscopic management of cholecysto-duodenal fistula after spontaneous resolution of intestinal obstruction component of gallstone Ileus − case report and literature review

Summary

Gallstone ileus is a rare complication of gallstones and is a comparatively unique cause of intestinal obstruction. It involves the development of a cholecysto-enteric fistula through which a gallstone can pass into the gastrointestinal tract. Spontaneous resolution of intestinal obstruction in gallstone ileus is extremely rare. We report a 71-year-old patient who presented with right hypochondrial pain for four months. She had a three-day history of absolute constipation and abdominal distention two months before presentation that resolved spontaneously. Computed tomography revealed pneumobilia. Laparoscopic exploration showed a cholecysto-duodenal fistula that was divided, the opening in the first part duodenum was closed and cholecystectomy was completed successfully. The presence of pneumobilia in a patient with gallstones should raise the suspicion of bilio-enteric fistula. The obstruction component of gallstone ileus can resolve spontaneously in rare occasions. Single stage laparoscopic management of cholecysto-duodenal fistula is safe and feasible in the presence of an experienced laparoscopic surgeon.

Keywords:

gallstone ileus – pneumobilia − intestinal obstruction − cholecysto-duodenal fistula − laparoscopy

INTRODUCTION

Gallstone ileus is a comparatively rare trigger of mechanical intestinal obstruction, it occurs in 0.15%–1.5% of cholelithiasis cases and <0.1% of ileus cases overall [1,2]. The most accepted explanation for such a rare clinical entity is the migration of a stone from the gallbladder through a cholecysto-enteric fistula to be impacted distally in the gut [3]. Gallstone ileus commonly affects elderly patients with cholelithiasis and can have a high rate of complications [4].

Surgical intervention for gallstone ileus involves both stone extraction to relieve the obstruction with or without closure of the fistula. Whether surgery should be completed as a one-stage or two-stage operation remains a subject of debate [5]. Spontaneous resolution of bowel obstruction caused by impacted stone is extremely rare [6]. Fistula can resolve spontaneously but its persistence is a possible aetiology for retrograde cholecystitis or gallbladder cancer [7−10].

Herein, we present a case of an elderly female who had a gallstone ileus and was successfully managed by a one-staged laparoscopic approach.

CASE REPORT

A 71-year-old woman presented electively to the hepatobiliary surgery unit in Alexandria Main University Hospital with recurrent attacks of right hypochondrial pain and occasional vomiting for four months with a history of absolute constipation and abdominal distention two months ago that lasted for three days and resolved spontaneously. She had a history of hypertension, asthma, and diabetes mellitus with no history of previous abdominal surgery. Her clinical examination revealed a mild right hypochondrial tenderness and small partially irreducible paraumbilical hernia that the patient was known to have for about twenty years.

Abdominal ultrasonography showed a contracted gallbladder over an echogenic shadowing stone. Complementary computerized tomographic scan (CT) showed pneumobilia with no intra- or extra- hepatic biliary dilatation and with normal caliber of small and large intestine. Laboratory studies including liver function tests, markers of biliary stasis and inflammatory markers were essentially unremarkable.

A cholecysto-enteric fistula was suspected in view of the presence of pneumobilia after ruling out other possible other etiologies, such as endoscopic retrograde cholangio-pancreatography (ERCP). The decision was to perform a diagnostic laparoscopy by one of the experienced hepatobiliary laparoscopic surgeons using the conventional four trocar approach for laparoscopic cholecystectomy [11].

Laparoscopic exploration revealed a fistula between the body of the gallbladder and the first part of the duodenum (Fig. 1). The fistula was disrupted (Fig. 2) and the duodenal opening was closed in double layers (Fig. 3). That was followed by an uneventful laparoscopic cholecystectomy with a thorough irrigation of the abdomen and insertion of a drain (18 F) into Morison’s pouch. At the end of the procedure, the paraumblical hernia was reduced, the defect was closed and an onlay prolene mesh was fixed to the anterior rectus sheath. The patient had a smooth postoperative course and was discharged on the 5th post-operative day.

DISCUSSION

Bilio-enteric fistula is a rare complication of cholelithiasis with an estimated incidence of less than 5% [12]. Specifically, cholecysto-duodenal fistula (CDF), first described by Courvoisier in 1890, represents the most common type (80%) of bilio-enteric fistula which also includes cholecysto-colonic, cholecysto-gastric and cholecysto-jejunal, in order of frequency. CDF usually affects elderly females with other comorbidities [13].

The pathogenesis of CDF is justified by two theories: repeated attacks of cholecystitis leading to adhesions between the gallbladder and the duodenum with consequent erosion and fistulation or pressure necrosis of the gallbladder by a large gallstone. Other causes of CDF include peptic ulcer, trauma, malignancy and inflammatory bowel disorders [14].

After development of CDF, gallstones can freely gain access to the gastrointestinal tract and CDF has a spectrum of clinical presentations based on the gallstone size. The full-blown picture is gallstone ileus (GSI) with complete intestinal obstruction [14]. CDF may be asymptomatic if the gallstone size is less than 25 mm with unnoticed passage. It can also be an incidental finding during an elective cholecystectomy, or cause recurrent attacks of ascending cholangitis [14]. Gallbladder cancer can be a complication in 0.82% of CDF patients [15]. Rarely, GSI may present with recurrent attacks of partial intestinal obstruction indicating movement of gallstones. Some patients may complain of vague non-specific symptoms such as abdominal pain, diarrhea and steatorrhea.

Diagnosis of CDF is challenging owing to the absence of specific symptoms, signs, laboratory markers or radiological findings. Preoperative diagnosis is achieved in only 17% of patients; however, it is crucial to avoid unplanned procedures and undesirable preventable complications. Rigler’s triad, first described in 1941, is pathognomonic for GSI and consists of intestinal obstruction, pneumobilia (aerobilia), and ectopic gallstones [16]. Intestinal obstruction is reported in 70% of patients with bilio-enteric fistula [17].

Pneumobilia, the most common radiological finding associated with CDF, is detected in one third of these patients. Radiologically, the presence of pneumobilia is always abnormal and, hence, may provide the first clue of the bilio-enteric fistula. However, it may be attributed to other pathologies such as emphysematous gallbladder, hepatic abscess, or portal vein gas. It can also be seen in patients with sphincter of Oddi dysfunction or after biliary tree intervention such as ERCP [13,18]. Trans-abdominal ultrasonography is not accurate in detecting CDF, however, it could raise the suspicion for bilio-enteric fistula and indicate further imaging as in the case of a contracted gallbladder with absence of previously-noted gallstones [12]. Magnetic resonance cholangiopancreatography (MRCP) provides further data about biliary tree anatomy but is only indicated in certain situations.

There are ongoing discussions regarding the best management approach for CDF, but it mainly depends on its clinical presentation. For GSI, the most common presentation, the standard management is surgery without a consensus on the best approach: one-staged versus two-staged approach [16]. Furthermore, some authors have reported a successful conservative trial with spontaneous resolution of GSI [6,17,19]. Incidentally- discovered CDF can be feasibly managed as a onestaged procedure including cholecystectomy and fistulectomy or fistula repair depending on good general condition and presence of an experienced hepatobiliary surgeon. Therefore, if the patient’s general condition is poor and/or the surgeon is not skilled or experienced, it is advised to postpone dealing with the fistula until the general condition of the patient improves and an experienced hepatobiliary surgeon is made available.

Laparoscopic management can be safely performed with experienced hands and should be performed in a well-equipped specialized center [5]. Indeed, laparoscopic management of CDF is challenging especially in the presence of dense adhesions, surrounding inflammation, distorted anatomy, multiple fistulae, bleeding and technically-demanding laparoscopic intestinal suturing. Therefore, the risk of vascular and biliary injury is higher. A conversion to open surgery is expected and has been frequently reported [12,13]. However, conversion from laparoscopy to open surgery has declined overtime owing to the improved surgical techniques and building experience in the field of laparoscopic hepatobiliary surgery [20].

There are multiple interesting points in our case. The first point is her clinical presentation. She was an elderly lady with multiple comorbidities and presented with recurrent typical biliary colic. However, her ultrasonography showed a contracted gallbladder and her abdominal CT revealed pneumobilia. She had no other abnormal or suspicious laboratory or imaging findings. At this point, an incidental finding of CDF was assumed, though, the complete history and workup were not enough to establish a preoperative diagnosis of CDF. The second interesting point is the spontaneous resolution of her gallstone ileus without diagnosis or intervention. Retrospectively and based on pneumobilia and presence of CDF, her transient intestinal obstruction was better attributed to CDF rather than obstructed paraumbilical hernia. The spontaneous resolution of GSI has been only reported in a few articles after a trial of conservative approach for otherwise-unfit patients [17].

The third interesting point is the management approach, based on the diagnostic laparoscopy, a clear fistula was found to connect the gallbladder and the first part of the duodenum. We opted to perform a laparoscopic one-stage procedure including cholecystectomy and fistula repair which was successfully completed, and the patient had a smooth uneventful postoperative course. We preferred the laparoscopic approach, based on our experience, to avoid the complications of laparotomy and its lengthy postoperative recovery.

CONCLUSION

Cholecysto-enteric fistula is a rare complication of cholelithiasis and the presence of pneumobilia in a patient with gallstones should raise suspicion of a fistula between the biliary tree and gut. High index of suspicion, based on detailed history and imaging findings, is important for its preoperative diagnosis. The obstruction component of gallstone ileus can resolve spontaneously in rare occasions. Single stage laparoscopic management is a feasible and safe approach when performed by an experienced hepatobiliary laparoscopic surgeon. Despite its rarity, gallstone ileus can be unnoticedly resolved.

Ethics approval: Not applicable for case reports according to the local policies of Alexandria University.

Consent: The patient signed an informed consent and it is available upon request.

Conflict of interests

The authors declare that they have not conflict of interest in connection with this paper and that the article has not been published in any other journal, except congress abstracts and clinical guidelines.

Mohammed S. Foula

Department of Surgery

King Fahd Hospital of the University,

Imam Abdulrahman Bin Faisal University

e-mail: mohamed.foula@gmail.com