After initial signs of recovery, our patient developed worsening abdominal pain on the fourth post-operative day, and abdominal CT was undertaken (Figure ​(Figure1).1). The scan revealed a low-grade small bowel obstruction due to a laminated ileal gallstone, a fistula between the gallbladder and duodenum, and a further stone in the gallbladder. After a trial of conservative management, a second laparotomy on day nine revealed a gallstone impaction in the distal small bowel and a contained leak in the original ileal anastomosis. The stone was removed through a transverse enterotomy, and the anastomosis was refashioned with sutures. The inflammatory mass in the right upper quadrant, with the residual gallbladder stone, was palpable but not disturbed. The post-operative recovery was complicated by a Clostridium difficile infection, but our patient returned home to independent living four weeks after the second procedure.

Twelve months later, she presented with a further episode of central abdominal pain associated with vomiting and distension. An ultrasound scan revealed a contracted gallbladder with a 10mm common bile duct, and intravenous antibiotics were commenced for a presumed cholecystitis. She failed to settle, and a CT scan revealed free intraperitoneal gas and fluid, a persistent cholecysto-duodenal fistula, and a further gallstone obstructing the small bowel. This gallstone corresponded in size and shape to that seen in the gallbladder 12 months earlier (Figure ​(Figure2).2). At laparotomy, a limited small bowel resection was performed for perforation proximal to a gallstone obstruction. The stone was removed, and a hand-sewn anastomosis was performed. Her post-operative recovery was again complicated by a C. difficile infection in addition to a prolonged period of respiratory failure managed with a tracheostomy in critical care. She was discharged to her own home six weeks after surgery and made a good recovery. Two months after discharge, magnetic resonance cholangiopancreatography did not visualize any residual gallbladder stones.

Operative management is challenging. Simple enterotomy risks further stones and the potential complications of cholangitis, pancreatitis, and recurrent gallstone ileus. Conversely, cholecystectomy and fistula repair within dense adhesions risk duodenal injury and biliary leak. Both options have an associated mortality in the population in question.

In the case described, it is unclear whether the stone removed at the second laparotomy was in the gallbladder or the bowel lumen at the time of the first operation. Twelve months later, the CT images suggest that a further stone had migrated into the bowel. Our experience demonstrates the importance of inspecting the entirety of the small bowel at the time of the initial laparotomy and illustrates that late recurrence of gallstone ileus, albeit rare, can occur [5].

To the best of our knowledge, this is only the third reported case of twice recurrent gallstone ileus [6,7]. The recent timing of the two previous reports suggests that the phenomenon is becoming more common, presumably as a result of the increasing age of the population, and prompted us to revisit the evidence comparing enterotomy alone with the one-stage approach in the initial management of the condition.

To date, the largest review of reported cases of gallstone ileus, which consisted of 1001 cases, was published by Reisner and Cohen in 1994 [1]. The findings are frequently quoted, but the work is limited by the heterogeneity of the reports analyzed. We undertook an electronic literature search to identify all subsequent published comparisons of enterotomy alone versus one-stage repair in the management of gallstone ileus. The terms “gallstone ileus” and “gallstone obstruction” were inserted into PubMed, reference lists were checked, and Scopus (ScienceDirect) was used to identify the citations of key articles. We identified eight comparative studies, of which three comprised at least five patients in each arm. The smaller studies were excluded. Three primary endpoints of operative mortality, operative morbidity, and recurrent biliary disease were considered. Operative complications were defined as those preceding discharge from hospital. Recurrent biliary disease is defined as cholecystitis, choledocholithiasis, cholangitis, recurrent gallstone ileus, pancreatitis, or cholangiocarcinoma during follow-up. The findings of the three studies identified, in addition to those of Reisner and Cohen, are presented in Table ​Table1.1.

In their combined series, Reisner and Cohen [1] published operative mortality rates of 16.9% for one-stage repair and 11.7% for enterolithotomy (P <0.17). The three more recent series also suggest a non-significant excess in operative mortality following one-stage repair. Where reported, comparison of morbidity between the two procedures appears to favor enterotomy and this finding was statistically significant in the study by Doko et al. [8]. There are no data comparing one- or two-stage repair with simple enterotomy in the context of recurrent gallstone ileus. Interestingly, none of these papers reported an increased frequency of recurrent biliary disease at follow-up. They do, however, illustrate two important points: the high levels of comorbidity in this patient group and the increased operating times associated with one-stage repair [8,9].