AIM: To help the surgeon in decision making when treating a patient with recurrent gallstone ileus (RGSI).

METHODS: A systematic review related to RGSI was performed using the databases CINAHL, EMBASE, MEDLINE via PubMed from May 1912 to April 2015. All languages were included and the grey literature was also searched. The abstracts were explored for relevance to the topic and full texts obtained as appropriate. A manual search was carried out by scrutinising the reference lists of all the full text articles and further articles were identified and obtained. Total of 903 articles were identified, 656 were excluded after abstract review, 247 full text articles were reviewed and 91 articles selected for final analysis. There were 113 cases of RGSI.

RESULTS: There were 113 cases of RGSI reported in 91 articles. The majority of the recurrences, 62.6%, occurred within 6 wk of the index event. The male to female ratio was 1:7. The mean age was 69.6 years (SD 11.2) with a range of 38-95 years. The small bowel was the commonest site of impaction (92.2%). Treatment data was available for 104 patients. The two main operations performed were: (1) Enterolithotomy without repair of biliary fistula in 70.1% of all patients with a procedural mortality rate of 16.4% (12/73) and (2) a single stage surgery approach involving enterolithotomy with cholecystectomy and repair of the biliary enteric fistula in 16.3% with a procedural mortality of 11.7% (2/17). A subset analysis over last 25 years showed mortality from eneterolithotomy was 4.8% while single stage mortality was 22.2%. Enterolithotomy alone was the commonest operation performed for RGSI with four patients (5.4%) having a further recurrence of gallstone ileus.

CONCLUSION: Enterolithotomy alone or followed by a delayed two-stage treatment approach is the preferred choice offering low mortality and reduced risk of recurrence.

Gallstone ileus (GSI) is a rare entity first described in 1654 by Bartholini[1,2]. GSI is a result of an inflamed gallbladder becoming adherent to an adjacent part of the enteric system and discharging its stones into the enteric lumen through formation of a biliary-enteric fistula[3-5]. It is estimated that 80% of intraluminal stones will pass spontaneously[6], Gallstones measuring more than 2.5 cm in diameter are a risk factor for impaction and causing bowel obstruction[7]; the latter is referred to as GSI. The mainstay of management is surgical treatment involving enterolithotomy alone or combined with a cholecystectomy and repair of biliary-enteric fistula as a single stage procedure.

Recurrent gallstone ileus (RGSI) is usually a consequence of an untreated biliary-enteric fistula with cholelithiasis[8]. An alternative hypothesis is the presence of a non obstructive biliary calculus more proximal in the small intestine which escaped detection at the first operation despite the need to palpate the entire small bowel looking for a second stone[9]. Predicting the risk of RGSI at the time of first operation is difficult. The literature reports an estimated risk of RGSI of 5%-8%[10-12].

When a patient presents with RGSI the surgeon will not only have to consider how to deal with the emergency obstruction but also how best to manage the cause of the recurrence. There are advocates for enterolithotomy alone without dealing with the biliary-enteric fistula, as low morbidity and mortality are perceived to be associated with this approach. However the advantages of repairing the fistula include preventing recurrence, ascending cholangitis and gallstone related complications[13-15]. These issues are similar to the ones at primary presentation of Gall Stone ileus but increase in significance now as at the primary presentation the risk of recurrence is only 5%-8%.

Although there have been several reviews of GSI, there has been no review focusing on RGSI since 1998[11]. Following a case in our hospital where a patient presented with two recurrences of GSI[16], we performed an up to date systematic review to gain a better understanding of its presentation, management and outcomes. This review will assist clinicians with the management of this rare but important condition.

An electronic search was performed using CINAHL, EMBASE, MEDLINE via PubMed, from inception of each database to April 2015. A web-based search was also carried out using the Boolean Internet search engine “Google”. The search terms used were; “recurrent” or “recurrence” and “intestinal obstruction”, “gallstone” or “GSI”. The search included articles written in any language.

The abstracts were explored for relevance to the topic and full texts obtained as appropriate. A manual search was carried out by scrutinising the reference lists of all the full text articles and further articles were identified and obtained.

A search of the grey literature was undertaken by searching the Royal College of Surgeon’s website and a search of the grey literature database Open Grey http://www.opengrey.eu/. No further articles were identified.

Thirty-six articles of potential relevance in languages other than English were identified. All the articles were translated by native speakers in health related professions. The translations were independently reviewed by the authors before a decision was made about whether the papers were relevant for this review. Of the 36 articles identified, 20 were subsequently included.

The search identified 903 articles from multiple sources as described. Ninety one articles were eventually included (Figure 1). These included 54 individual case reports, 34 case series and 3 review articles[10,17,18]. There were 113 cases of RGSI documented in the 91 articles. Full data were not available on all the categories of interest. Consequently the denominator for each category varies. This is made explicit in the text.

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Figure 1 PRISMA flow diagram. RGSI: Recurrent gallstone ileus.

The literature reports an estimated risk of RGSI of 5%-8%[10-12,74]. However reporting of RGSI is probably underestimated because the figures are based on published case reports or series.

The management of RGSI presents a dilemma for the surgeon. Should one only deal with the presenting obstruction once more, in which case an enterolithotomy will suffice, or should one now also deal with the cause of the recurrence in which case additional cholecystectomy and repair of biliary-enteric fistula will be needed. We sought to review existing literature that would help clinicians choose appropriate treatment strategies when faced with RGSI.

In our review RGSI mainly occurred in patients who had their primary GSI treated with enterolithotomy (86.7%). However two patients who had RGSI had single stage surgery including biliary-enteric fistula repair at the initial episode[5]. The latter suggests that recurrence can be due to pre-existing stones in the bowel that have been missed. Identification of multiple stones at the outset is therefore likely to be helpful. While a pre operative CT scan may help, careful per operative manual searching for additional stones is crucial. The authors have personal experience with a patient who had two episodes of recurrent Gall Stone Ileus having been noted to have visible stones within the gall bladder on the CT scan at the time of initial and second presentation[16].

With regard to per operative searching for additional stones, the shape of the index stone may be a useful indicator. The presence of a faceted or a cylindrical stone at the time of first surgery suggests presence of multiple stones[10]. Most of the articles in our review did not comment on the shape of the stone but of the 36 articles that did, 83.3% of the stones were faceted. This suggests that a search for additional stones may be required more often than not, and that the search will be productive.

Treatment of RGSI is usually surgery, though our review found eight cases that had been dealt with conservatively. The mortality in these latter cases was very high (25%) and therefore should be avoided unless severe co-morbidities prohibit surgical intervention.

The surgical options include enterolithotomy alone with removal of the stone thus relieving the obstruction or an enterolithotomy with a definitive operation involving cholecystectomy and repair of the biliary-enteric fistula in order to prevent future RGSI.

Enterolithotomy alone is seen as technically less demanding than single stage surgery. The increased complexity of the latter procedure theoretically carries a higher operative risk. In addition, elderly patients with multiple medical co-morbidities may present a greater physiological risk and this has to be factored into the management of RGSI.

In our review of RGSI cases comparing the recent 25 years to the preceding 77 years, the operative mortality for single stage surgery was 22.2% (1990-2015) compared to 0% (1912-1989). This is despite advanced in surgical techniques and perioperative care. However these results must be interpreted with caution as this is based on published cases only and relatively small numbers of patients.

Of the cases treated with enterolithotomy the mortality rate for 1912-1989 was 21.2% compared to a rate of 4.8% for 1990-2015. The latter concurs with the mortality rate of 5% reported in 2013 by Halabi et al[93] from their analysis of the Project Nationwide inpatient sample (NIS) database of just over 2000 cases.

The mortality rate of the whole cohort over the last 100 years was lower for single stage surgery in comparison to enterolithotomy despite the procedure being technically more demanding (11.7% vs 16.4%). If age is used as a surrogate marker for physiological fitness then we can perhaps assume that patients undergoing single stage surgery were not only younger but also fitter. However the number of patients having single stage definitive surgery was small and data on age was not available for all patients therefore caution must be taken in the interpretation of these results.

A two-stage strategy with initial enterolithotomy followed by an elective cholecystectomy and biliary-enteric fistula repair had a better outcome with 0% mortality in the seven patients[10,17,39,51,55,59,83], however this represents less than 10% of the cases and probably represents a selection bias in patients fit enough to consider an elective second surgery. However this option should also be considered in the management of RGSI.

The mortality rates from enterolithotomy alone have reduced in the last 25 years and there is a risk, albeit low, of further recurrence. We recommend it as an appropriate choice for the management of RGSI especially for the non hepato-biliary surgeon who has to deal with an emergency obstruction caused by RGSI.

To deal with the problem of possible recurrence Single stage definitive surgery, despite being more technically demanding, may be worth considering but mortality rates remain high. This approach may be appropriate in younger patients who pose a lower risk. This concurs with recommendation from other authors who have reviewed the outcome of primary GSI.

With improvement of surgical techniques and perioperative care a delayed two-stage treatment approach may provide the best results in selected cases.

The authors wish to acknowledge the contribution of Christian Körner of University Hospital Giessen and Marburg, Marta Kankova and Sarah Braungart of York Teaching Hospital for help with translation of the Non- English articles.