Complete removal of the diseased colon and rectum is often necessary for cure in conditions such as ulcerative colitis (UC), Crohn’s proctocolitis, and indeterminate colitis. Fortu- nately, patients undergoing total proctocolectomy (TPC) today are almost always able to maintain intestinal continuity and an anatomical route for defecation through creation of an ileal pouch–anal anastomosis (IPAA). Before the 1970s, however, this was not the case. Patients of that era had an end ileostomy as the only option and the inconvenience and stigma attached to the ileostomy often made patients reluctant to consent to needed surgery. In 1969, a “continent ileostomy” (CI) was first described by Kock1 of the University of Gothenburg in Sweden, and was heralded as a major improvement over the conventional end ileos- tomy. The perceived advantages of the CI over an end ileostomy were derived from the intussuscepted valve that allows for creation of a stoma that is flush with the skin and does not require the patient to wear an external appliance to collect intestinal waste. A number of inflammatory bowel disease (IBD) centers, including

Received for publication June 10, 2014; Accepted June 29, 2014.

From the Department of Colorectal Surgery, Digestive Disease Institute, Cleveland Clinic, Cleveland, Ohio.

E. Aytac is supported by the Ed and Joey Story Endowed Chair in Colorectal Surgery. The other authors have no conflicts of interest to disclose.

Reprints: David W. Dietz, MD, Clinic Foundation, 9500 Euclid Avenue/A30, Cleveland, OH 44195 (e-mail: dietzd2@ccf.org).

Copyright © 2014 Crohn’s & Colitis Foundation of America, Inc. DOI 10.1097/MIB.0000000000000160
Published online 12 September 2014.

the Cleveland Clinic, adopted the procedure and began to gain experience with it during the ensuing decade. The description of the restorative proctocolectomy (IPAA) in 1978 by Parks and Nicholls,2 however, quickly displaced the CI from a position of prominence in the surgical treatment of patients with UC. Its demise was also abetted by the complexity of the operation and attendant high complication rates. As time passed, fewer and fewer centers offered CI as an option for patients with medically refractory UC and today only a handful of surgeons familiar with the procedure can be found in practice.

However, despite the fact that the CI is no longer a common procedure, it still has a role in the surgical management of patients with IBD, and physicians caring for IBD patients should have some familiarity with the procedure so that they may counsel their patients appropriately. This review will discuss the present day indications for CI surgery in patients with IBD, evolution of the modern CI, current surgical technique, and reported outcomes.

INDICATIONS FOR CI IN THE IPAA ERA

Today, IPAA has become the gold standard procedure for the surgical treatment of patients with UC.3 Within the IBD population, however, CI remains an option in certain specific situations in patients with ulcerative/indeterminate colitis and in very highly selected pa- tients with Crohn’s colitis.4 In our experience of 423 patients under- going CI surgery at the Cleveland Clinic, the final diagnosis was UC in 74%, indeterminate colitis in 5%, and Crohn’s colitis in 10%. Familial adenomatous polyposis accounted for the remaining 7%.

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There are 4 common situations where IBD patients in our institution may be offered a CI: (1) Patients who are referred for redo-IPAA after failure of their index IPAA and are found not to be candidates for that procedure; (2) patients who are taken to the operating room for restorative proctocolectomy in whom IPAA cannot be achieved because of inadequate mesenteric length; (3) patients who require TPC but have contraindications (e.g., incontinence, low rectal cancer, and anal canal disease) to IPAA; and (4) patients with an existing conventional ileostomy who find it unacceptable due to severe pouching problems or interference with sex life, athletics, or occupation. The 423 patients who had CI surgery performed in our institution as of 2012 followed these indications: conversion of a conventional end ileostomy to a CI (59%), TPC with CI (20%), conversion of a failed IPAA to a CI (16%), and completion proctectomy with CI (5%). Each of these indications will be discussed in detail below.

Failed IPAA

Failure of an IPAA is defined as the excision of the ileo-anal pouch, permanent diversion with a proximal loop ileostomy, or having an unreversed diverting ileostomy.5 The long-term risk of IPAA failure varies between 3% and 28% and is influenced by characteristics of the patient popula- tion.3,6 Pelvic sepsis complicating surgery and a diagnosis of Crohn’s disease (CD) are strongly associated with pouch failure.7,8

Patients with a failed IPAA are often candidates for redo- IPAA and good results can be obtained. In our experience with 241 patients undergoing an IPAA reconstruction, the overall success rate was 88%.5 However, a small percentage of patients who are referred to our institution for redo-IPAA are not candi- dates for that procedure. We consider factors such as poor sphincter function and extensive pelvic fibrosis due to peripouch sepsis as contraindications to redo-IPAA. Some of these pa- tients, however, can be offered a CI if it is felt that they will not do well with a conventional end ileostomy. Our experience with CI in 64 patients with a failed IPAA has been previously reported.9 In 25%, the existing J-pouch was able to be converted into a CI pouch, whereas 75% required excision of the existing pelvic pouch with creation of a de novo CI. The length of the remaining bowel is often an issue in these patients because the previously constructed pelvic pouch may comprise 15% of their existing bowel length. Because a CI requires approximately 55 to 60 cm of bowel for construction, a patient who undergoes excision of a failed pelvic pouch with creation of a new CI may lose up to one-third of their functional bowel length, as the absorptive capacity of the CI pouch is largely unknown. It is critical to consider and discuss with the patient the “worst case scenario” of subsequent failure of the CI that requires pouch excision. This would potentially leave the patient with only 200 cm of small bowel proximal to an end ileostomy, a bowel length that most would consider on the borderline of short bowel syndrome.

Technical or Patient-related Factors that Preclude IPAA After TPC

Occasionally, patients are taken to the operating room for restorative proctocolectomy but the surgeon finds that he or she is unable to create an IPAA because of inadequate small bowel mesenteric length. Patients requiring TPC may also be determined to be poor candidates for IPAA due to preexisting problems such as poor sphincter function, low rectal cancer complicating chronic colitis, or anal canal/perineal CD. In these latter situations where preoperative discussion has been held and consent obtained, a CI can be constructed at the same operation. However, in the former case, especially if not anticipated and discussed before surgery, it is best to construct a conventional end ileostomy. Discussions can then be held with the patient after surgery, and the conventional ileostomy can be converted to a CI 6 to 12 months later.

Existing Conventional Ileostomy Unacceptable

The largest group of patients undergoing CI surgery in our institution are those who present to us with an existing conventional end ileostomy. These patients seek a CI for a variety of reasons, but the common denominator is that they feel that their existing ileostomy is having an adverse effect on their quality of life. Common concerns relate to the effect of the ileostomy on sex life and limitations on physical activity, especially in young athletes. A small group of patients have skin conditions such as psoriasis or allergic dermatitis related to the stoma appliance. These patients have great difficulty in maintaining a seal and suffer from frequent and embarrassing leakage. In addition, the need for frequent pouch changes, sometimes several daily, can be financially prohibitive. Patients seeking CI for the above reasons should be counseled extensively. The risks of CI surgery, both short-term and long-term, must be balanced against the patient’s expectations and the likely benefits.

EVOLUTION OF THE CI

After first being described by Kock1 in 1969, the technique for CI creation has evolved through a number of modifications.10,11 Kock’s initial description of the CI did not include an intussuscep- ted nipple valve. The primitive design was a U-shaped pouch con- structed from the distal small bowel with a long efferent limb pulled through an opening in the abdominal wall within the confines of the rectus abdominis muscle and terminating in a skin-level stoma.1,11 The rectus abdominis muscle was intended to act as a sphincter-type mechanism around the efferent limb to provide continence.12 Unfor- tunately, this design only provided continence in a small minority of patients. Kock’s initial, albeit unsuccessful, modification to over- come the problem of incontinence was to create an antiperistaltic efferent limb. It was not until 1973 that he described intussusception of the efferent limb to create the characteristic nipple valve that is the hallmark of most modern continent ileostomies.11–15 This mod- ification was successful in providing continence to a majority of patients, but it also set the stage for the most common complication of the modern CI, which is valve slippage.16–20

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Over the past 30 years, a number of new methods have been developed in an attempt to reduce the rate of valve slippage, which has been reported in as many as 30% of CI patients.4,10,21,22 Kock attempted to address this problem through modifications of his technique that involved splitting and defatting of the valve mesentery, suture fixation and serosal scarring, partial rotation of the valve, and finally stapled fixation with the use of a fascial strip or synthetic mesh. Kock’s largest published series of 314 patients showed a steady reduction in valve complications and slippage with the evolution of his technique.23 Others have made similar modifications to the procedure, mostly aimed at fixation of the valve by chemical or physical means. Fibrosis between the 2 intussuscepted layers has been promoted by traumatizing the serosa of the efferent limb using an orthopedic rasp,24 deep dia- thermy scarring of the serosa,25 interposing synthetic mesh between the valve layers,26 and by chemical means with substan- ces such as formalin, silver nitrate, talc, and even asbestos.27 Staple fixation of the valve, both to itself and the pouch sidewall, was first described by Fazio and Tjandra28 in 1992, and this remains as the primary means of valve stabilization in our insti- tution today. Although these modifications have undoubtedly reduced the incidence of valve slippage, they have also increased the risk of other complications such as valve or pouch fistulas.29,30

More radical attempts at altering the basic design of the CI have also been undertaken in an attempt to decrease the risk of valve slippage. The most common of these is the Barnett continent ileostomy reservoir (BCIR). The initial form of the BCIR was described by Spencer and Barnett31 in 1979 and relied on an isoperistaltic intussuscepted valve for continence. However, valve slippage continued to occur, and the “living intestinal col- lar” that is the distinguishing feature of the modern BCIR was added in 1986 in an attempt to buttress the mesenteric side of the valve where slippage is felt to first develop.21

Regardless of the technique of CI construction, valve slippage remains as the “Achilles heel” of the operation.4,21,32 In an attempt to eliminate the problem of valve slippage altogether, Kaiser et al33 have developed a “valveless” CI. This design, known as the “T-pouch,” was initially described by Stein et al34 for urinary diversion after cystectomy. Although valve slippage is inherently avoided, analysis of the designers’ first 10 years of experience with the technique still found a reoperation rate of greater than 50%.22

CURRENT TECHNIQUE OF CI CREATION AT CLEVELAND CLINIC

Our technique of CI construction has been previously described.4,18 In brief, preoperative preparation includes stoma marking by our enterostomal therapists with a site typically cho- sen in the right lower quadrant within the confines of the rectus muscle. A CI is sited lower than a conventional end ileostomy, usually at the same level as the anterior superior iliac spine to account for the fact that the pouch will reside internally within the pelvis or lowermost aspect of the right lower quadrant abdominal

cavity (Fig. 1). The CI operation can be divided into 4 stages: construction of the S-pouch, creation of the intussuscepted nipple valve, valve fixation, and siting of the pouch with stoma matura- tion. In the case of de novo CI creation, the entire pouch is con- structed from the distal-most 60 cm of small bowel. In stage 1, an S-pouch is fashioned from three 12-cm to 15-cm limbs of ileum. These limbs are first approximated with a serosal layer of inter- rupted or continuous 3-0 polyglactin suture leaving an efferent limb of approximately 20 cm that will ultimately become the intussuscepted nipple valve. The exact length of the efferent limb is derived from doubling the desired length of the valve (6–7 cm) and adding the thickness of the abdominal wall through which the exit conduit will pass (Fig. 2A). An antimesenteric enterotomy is then created along the 3 limbs of the S-pouch (Fig. 2B), and the back wall of the pouch is constructed with a running full- thickness 3-0 polydioxanone suture (Fig. 2C, D). A 6- to 7-cm valve is then fashioned by intussuscepting the efferent limb into the pouch and fixing it to itself with 2 firings of a noncutting 55-mm linear stapler placed along either edge of the valve mes- entery (Fig. 2E, F). Care must be taken to not include the mes- entery in the staple lines as a hematoma or valve ischemia could result. If the efferent limb that will be used for valve construction has a bulky mesentery, it should be stripped of peritoneum and fat, taking care not to damage the underlying blood vessels. This “defatting” maneuver will make it easier to intussuscept the effer- ent limb to create the valve and will also promote fibrosis between the valve layers to inhibit slippage. The anterior wall of the S-pouch is then closed with either a running 3-0 polydioxanone or interrupted 3-0 polyglactin seromuscular suture(s). Suture closure of the anterior pouch wall is initiated at the apex of the

FIGURE 1. Immediate postoperative picture of a patient whose con- ventional ileostomy has been converted to a CI. The CI is intubated with a drainage catheter that will remain in place for 1 month to provide continuous drainage while the pouch heals. Note that the CI is sited below the level of the former conventional end ileostomy (open wound). The left lower quadrant drain terminates in the pelvis and will be removed before discharge of the patient from the hospital.

pouch, and each stitch includes the mucosa and submucosa of the antimesenteric aspect of the valve (Fig. 2G). This is important for fixation of the valve to the pouch wall, a maneuver that helps to minimize the risk of valve slippage. When the suture line reaches the tip of the valve, a third firing of the noncutting 55-mm linear stapler is applied along this suture line to further reinforce the suture fixation of the valve to the pouch wall (Fig. 2H). Stitches are then transitioned to only include the pouch wall, and the suture line is completed to close the anterior pouch wall. “Fundoplication” stitches of 3-0 poly(ethylene terephthalate) are then placed between the apex of the pouch and the exit conduit to further stabilize the valve. Pouch suture line integrity and continence of the valve are tested by in- tubating the pouch with a drainage catheter, filling the pouch to capacity with saline and air, and then withdrawing the catheter. The pouch is then situated within the pelvis or lower abdominal cavity, and the exit conduit is brought through the abdominal wall after creating a stoma aperture at the previously marked site. The apex of the pouch is then fixed to the underside of the abdominal wall with several 3-0 poly(ethylene terephthalate) sutures. The stoma is again intubated with the drainage catheter, the location of its tip is confirmed within the pouch, and the catheter is secured to the skin to prevent it from becoming dislodged in the postoperative period. The pelvis is drained and the abdomen is then closed.

The drainage catheter is left in place for 4 weeks after surgery to keep the pouch continuously drained, using frequent

small-volume irrigations to ensure patency. The first outpatient visit occurs 1 month after surgery, the time at which the CI is tested for continence and capacity. Intermittent catheterization begins at 2 hourly intervals, with a gradual reduction in the frequency of intubation over the ensuing weeks. Routine follow- up is scheduled at 3 months from the discharge day and at yearly intervals thereafter. Pouchoscopy is performed at those visits to assess the valve for signs of slippage (Fig. 3).

POSTOPERATIVE COMPLICATIONS AND LONG- TERM OUTCOMES AFTER CI

Postoperative complications are common after CI sur- gery,4,18,29 a fact that has likely restricted its widespread use. The procedure is safe, however, with reported mortality rates that are similar to any major abdominal operation.4–6 Complications occurring within the first 30 days after surgery include wound infections, staple-line bleeding, suture line leaks and enterocuta- neous fistulas, and valve necrosis. The latter is exceedingly rare (1 occurrence in the senior author’s [D.W.D.] 13 years of expe- rience), but would likely require pouch excision with immediate conversion to a conventional ileostomy. In select cases, the valve only could be excised, the resulting pouchotomy closed, and a proximal diverting loop ileostomy created. This would allow for the possibility of eventual pouch salvage, with creation of

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Continent Ileostomy in Inflammatory Bowel Disease

FIGURE 3. Endoscopic view of a normal CI nipple valve 1 year after surgery. The ileoscope has been introduced into the pouch and ret- roflexed to view the valve. Note the normal valve length of 6 to 7 cm and the fixation of the valve tip to the pouch sidewall by a mucosal bridge. This is the end result of the staple fixation maneuver illustrated in Figure 2H.

a new valve from the afferent limb of the pouch 6 to 12 months later. Enterocutaneous fistulas after CI surgery often close spon- taneously if the pouch is kept to prolonged continuous drainage. These patients are usually managed with total parented nutrition and nothing by mouth until healing occurs. Staple-line bleeding is

managed conservatively with supportive care and transfusion of blood products and coagulation factors as needed. The pouch should be irrigated frequently to remove clots. If bleeding fails to cease, then endoscopic management is undertaken. An endo- scopist experienced with the anatomy of the CI is best suited to deal with this difficult situation.

Long-term outcomes after CI surgery have been reported in several large series (Table 1), with the authors generally concluding that although reoperations are common, the majority of patients can be maintained with a well-functioning CI for many years.4,20,23,37,41 Valve-related complications including valve slippage, fistula, and prolapse are the common CI complications requiring revision sur- gery, whereas other more mundane problems such as pouchitis and bacterial overgrowth are managed medically. Parastomal hernias occur frequently and, contrary to those arising around conventional stomas, should be repaired when discovered as they contribute to the mechanism of valve slippage.4

As previously stated, valve slippage is the “Achilles heel” of the CI and is responsible for the majority of reoperations in CI patients.4,23,29,41 The first sign of a slipped valve is usually the dif- ficulty with intubation of the pouch. This occurs due to the angu- lation of the exit conduit that results as a portion of the intussuscepted valve slips and moves into the subcutaneous space. The patient may also notice mild prolapse of the stoma at this time. If valve slippage progresses, incontinence to gas and stool will follow as the intrapouch segment of valve is no longer adequate to close with rising pouch pressures. Patients may first notice incon- tinence in the morning when pouch pressures are highest after

First Author

Mullen et al21
Nessar et al4 Handelsman et al35 LepistÖ and JÄrvinen36 Litle et al37 Berndtsson et al38

Lian et al9
Wasmuth and Myrvold39

Behrens et al40 Kaiser22

Unpublished CCF data

n Year

510 1995 216 2006 100 1993

96 2003 85 1999 68 2004

64 2009 63 2009

42 1999 40 2011

423 —

Overall Revision, %

21

—

11 59 45 65

45 44

43 30

72

Most Common Complicationa

Valve slippage
Valve slippage Incompetent valve Nipple valve dysfunction Valve dysfunction Nipple valve dysfunction

and/or fistula Difficult intubation and

valve leakage
Nipple valve sliding and/or

pouch loosening

Slipped valve, fistula

Skin-level stoma complications

Nipple valve slippage

Failure, %

7 22 17 24 40 6

5 10

5 10

21

Mortality, nb

0 1 0 2 0 —

0 0

0 0

1

Follow-upc

2, mean 11, median 2.5d
18, median 11.4, mean 31, median

5, median

12, mean

3.4, mean 6.2, median

16, median

Study Period 1988–1991

1977–2001 1975–1989 1972–2000 1975–1995 1967–1974

1982–2007 1983–2007

1989–1996 2000–2010

1973–2011

Aytac et al

Inflamm Bowel Dis  Volume 20, Number 12, December 2014

a night’s sleep without intubation. In our published series of 330 patients undergoing CI surgery from 1975 to 2001, the overall reoperation rate was 70%.4 This rate is similar to reports from other high-volume experiences.23,41 However, after a median follow-up of 11 years, 10- and 20-year pouch survival rates were 87% and 77%, respectively, and the median length of pouch survival was 27 years. Maintenance of a functioning pouch came at the cost of an average of 2.9 reoperations per patient with a median revision-free interval of only 14 months. These data suggest that when a patient develops a slipped valve, they are likely to have problems with recurrent valve slippage over the lifetime of their pouch. Patients whose valve does not slip within the first several years after CI creation are likely to remain without the need for reoperation. A thorough discussion of surgical techniques to correct a slipped valve is beyond the scope of this review. Suffice it to say that these operations are technically demanding and often require considerable creativity on the part of the surgeon. Operations to address CI complications should only be undertaken by an experienced CI surgeon, as on several occasions we have seen patients whose CIs could have been salvaged, but were instead excised before referral.

In our experience, 20% of continent ileostomies ultimately fail and require conversion to a conventional end ileostomy. Predictors of CI failure are a diagnosis of CD (hazard ratio, 4.5), female gender (hazard ratio, 2.4), development of a fistula (hazard ratio, 3.0), and obesity.4 Weight gain is the greatest patient- controlled enemy of the CI valve, and patients should be counseled extensively before surgery regarding the need to maintain a healthy weight. We have found that the risk of pouch failure increases nearly 2.5 times for every 5-unit increase in body mass index.4 The other predictors of pouch failure are not able to be controlled.

QUALITY OF LIFE IN CI PATIENTS

The overall goal of CI surgery is to improve the patient’s quality of life compared with that which exists, or is anticipated to exist, with a conventional end ileostomy. As most reported series of CI surgery demonstrate, both short-term and long-term com- plication rates are high, and the majority of patients will require further surgeries during their lifetime to maintain acceptable func- tion of the CI pouch. It is imperative, therefore, to have reasonable evidence that CI patients do gain an improved quality of life compared with those with a conventional ileostomy, as the risks involved are considerable.

Several authors have examined quality of life in CI patients. Two studies compare quality of life in patients before and after conversion from a conventional end ileostomy with a CI. As might be expected, Ojerskog et al42 found that patient expect- ations, attitudes, and emotional reactions were “more positive” after CI surgery than just before. They also demonstrated improved working capacity after conversion to a CI and reported that the greatest effect was seen in leisure activities and quality of sexual life. Family and social relations, however, were not influ- enced by conversion to a CI. In our institution, quality of life measures for patients with a CI were found to be higher on all

scales in comparison to patients who had their CI removed and then reverted to a conventional ileostomy.4 The Cleveland Global Quality of Life score, a validated patient-rated tool for use in IBD, was significantly higher in the CI patients compared with those who reverted to a conventional stoma (0.87 versus 0.70; P 1⁄4 0.006).4 In our most recent examination of questionnaire data from nearly 200 patients undergoing CI surgery at the Cleveland Clinic, 95% stated that they would undergo CI surgery again and would recommend the procedure to another patient in need.

Two groups have compared quality of life between patients with a CI, conventional ileostomy, and IPAA. The Mayo Clinic found that their CI patients had fewer restrictions in sports and sexual activities compared with those with a conventional ileos- tomy, but had more difficulty with travel. As might be expected, patients with an IPAA had the fewest restrictions. No differences in social life, recreation, work, and family matters were seen between the 3 procedures.20 A study from the Netherlands by Hoekstra et al43 found that the overall quality of life in CI patients was neither significantly better nor worse than patients with either a conven- tional ileostomy or IPAA, although sexual enjoyment, gastrointes- tinal tract symptoms, and male sexual problems did differ between procedures. Nearly all patients were said to be “very satisfied” with the CI, and all stated that they would undergo the procedure again and would recommend it to others. Similar results have been found following the BCIR procedure, with patients enjoying improved quality of life, state of mind, and overall health.21

Continent Ileostomy in Crohn’s Disease
CD is generally considered as a contraindication to CI creation because the risk of pouch failure is significantly higher than in patients with UC.4,35 However, given our successful expe- rience with IPAA in highly selected patients with Crohn’s proc- tocolitis,44,45 we will offer CI if they are not candidates for IPAA. We will also offer CI to patients with CD who have previously undergone TPC and conventional ileostomy if they find their ileostomy to be unacceptable. This is a very highly selected group, however, with no history of small bowel disease and, preferably, a disease-free interval of at least several years after creation of their end ileostomy. We have not published our expe- rience with CI in patients with CD, but the situation is analogous to those with a known diagnosis of CD who undergo IPAA. The

10-year IPAA survival rate in this group is 85%.45

CONCLUSIONS

The CI retains an important, albeit small, role in the surgical treatment of patients with IBD. CI provides an alternative to the conventional ileostomy for patients who are not candidates for IPAA or who have an existing ileostomy that is adversely affecting their lifestyle. Despite a high reoperation rate related to valve complications, patients can maintain a well-functioning CI for many years that fulfills their expectations for enhanced quality of life. Providers caring for patients with IBD should be familiar with the procedure so that they may counsel their patients appropriately.

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