Perineal salvage surgery for prolapse of ileal pouch anal anastomosis: Systematic review and meta‐analysis

Abstract

Background

Ileal pouch anal anastomosis (IPAA) prolapse is a rare mechanical complication, with a paucity of literature on frequency and management. This study aimed to assess the prevalence and management outcomes of perineal salvage procedures (SPs) for IPAA prolapse.

Methods

Systematic search conducted in PubMed and Scopus with an additional search of relevant cross‐referenced literature on experimental and observational human studies involving IPAA for ulcerative colitis or familial adenomatous polyposis from database inception to March 2023. Risk of bias assessment was performed using the NIH quality assessment tool for observational cohort and cross‐sectional studies. Meta‐analysis was performed by standard methodology using a random‐effect model. Main outcome measure was the success rate of perineal SPs for pouch prolapse.

Results

Seventy‐two publications were screened; 3 observational studies met the inclusion criteria. Included studies comprised 27,061 patients who underwent IPAAs over a 37‐year study period. Ninety‐five patients had IPAA prolapse and pooled prevalence of 0.4% (95% CI: 0.3–0.4; I ² = 0%). Patient age ranged from 31.6 to 38 years with a male‐to‐female ratio of 1.2:1. Time to prolapse from primary IPAA construction was heterogeneously reported as 2.6 years [<2 years (48%), and ≥3 years (47%)]. Pouch advancement was most frequently reported (88.4%; 95% CI: 77.5–99.3). Overall morbidity and success rates were 23% and 90.5%, respectively.

Conclusion

Studies were heterogeneous. Perineal SPs were infrequently performed for the uncommon IPAA complication of prolapse, with good outcomes in mucosal prolapse. There is insufficient evidence on functional status and quality of life following perineal SPs for full‐thickness prolapse.

INTRODUCTION

Ileal pouch anal anastomosis (IPAA) surgery has evolved through technical innovations since its first description in 1978 [1, 2]. This reconstructive surgery is often the preferred surgical treatment for mucosal ulcerative colitis (MUC) and familial adenomatous polyposis (FAP). Long‐term durability of more than 90% has contributed to the increased popularity of IPAA [3, 4]. However, complications associated with IPAA may arise and require surgical treatment. These complications include sepsis, fistula formation, inflammatory, mechanical and functional disorders [5, 6, 7, 8]. Prolapse is a rare mechanical complication of IPAA. In a large retrospective review of patients who underwent IPAA, 0.3% of patients were diagnosed with pouch prolapse [9]. Pouch prolapse can be either mucosal or full‐thickness, akin to rectal prolapse [9]. There is a paucity of literature on the frequency and management of ileal pouch prolapse.

Reoperative surgery following IPAA is technically demanding but, in expert hands, can be performed with a high success rate [10, 11, 12]. The decision as to which technique to use for pouch salvage surgery is influenced by the specific indication [13]. While pouch excision and creation of an end ileostomy is the last resort treatment, salvage procedures (SPs) may still be attempted and are broadly divided into pure perineal repairs, abdominal or combined abdominoperineal operations [14, 15, 16]. Perineal SPs do not require entry into the abdominal cavity. They can be completed through a perineal, transanal, transvaginal or transgluteal route, whether or not assisted with endoscopic or imaging‐guided methods [14]. The perineal approach has a lower complication rate of approximately 20% compared to the transabdominal approach of 36%–61% as it avoids the inherent risks associated with pelvic dissection; however, it has a higher recurrence rate than abdominal SPs (50% vs. 15%) [17, 18, 19]. Generally, SPs are acceptable if postoperative morbidity is low, and both QoL and function are similar to those observed in patients with successful primary IPAA. This study aimed to assess the pooled prevalence and management outcomes of different perineal SPs for IPAA prolapse.

METHODS

This review was prospectively registered at the prospective register of systematic reviews PROSPERO (CRD42022373418) and reported according to the Preferred Reporting Items for Systematic Reviews and Meta‐Analyses (PRISMA 2020) statement [20]. Ethical approval was waived for the conduct of this study due to the nature of the data used.

Search strategy

A systematic search was conducted in PubMed and Scopus from the inception of each database to March 2023, with an additional search of relevant cross‐referenced literature. The search terms were as follows: (prolapse OR pouch failure OR floppy pouch) AND (ileal pouch anal anastomosis OR IPAA OR ileoanal anastomosis OR restorative proctocolectomy) AND (redo OR reoperation OR revision OR repair) AND (perineal OR transanal OR transvaginal OR transgluteal).

Eligibility criteria and study selection

The eligibility criteria included experimental and observational human studies that involved individuals with MUC or FAP who had undergone IPAA and subsequently experienced pouch prolapse. There were no restrictions based on the sample size and follow‐up of the studies. Studies that included paediatric patients (age <18 years) and those that purely assessed abdominal or combined abdominal and perineal SPs for pouch prolapse without a record of pure perineal SPs were excluded. Also excluded were case reports and meeting abstracts without full text. Independent research was conducted by two authors (ERO and ZG) with a consensus agreement on the publications for eligibility after the joint screening of titles and abstracts in a meeting. A resolution of any difference of opinion was made by the senior author (SDW). The reference lists of the articles initially found eligible were manually screened for additional eligible studies.

Data extraction

Data on authors' names, period of study, study design, population and setting, demographics, presenting symptoms and indication for IPAA were extracted. In addition, the time from pouch creation to prolapse, pouch configuration, method of anastomosis and redo repair method were captured. A histology of any resected specimen and relevant details on complications such as sepsis, wound healing, recurrence, functional outcomes and quality of life were extracted.

Risk of bias

The risk of bias assessment of the quality of individual studies was performed using the National Institutes of Health (NIH) quality assessment tool for observational cohort and cross‐sectional studies [21]. This assessment tool comprises 14 questions related to the quality of publication, with a summary assessment of ‘good’, ‘fair’ or ‘poor’ ratings.

Summary measures

Summary sheets were collated on Microsoft Excel for baseline characteristics of studies, the proportion of patients with pouch prolapse and demographics of affected individuals, including age. Clinical features of patients with pouch prolapse, including presenting symptoms, length of time to prolapse in months and indication for primary IPAA pouch configuration, were collated. The type of prolapse was categorized as partial thickness (mucosal) and full thickness pouch prolapse. The short (30‐day post‐operation), medium (>30 days to 1 year) and long‐term (>1 year) outcomes of the perineal SPs were summarized based on available data.

Outcomes

The primary outcome of interest was the success rate of perineal SPs for pouch prolapse. Secondary outcomes were the prevalence, complications and functional outcome (bowel function, urgency, incontinence, need for antidiarrheal medications, erectile function and quality of life) of perineal SPs.

Definition of terms

Pouch failure is defined as the need for pouch resection, permanent diversion or a redo pouch [22]. Successful ileoanal pouch salvage is defined as an intact functioning pouch after resolution of the problem, at a minimum follow‐up of 6 months [13]. Postoperative morbidity included any complication that occurred within 30 postoperative days.

Statistical analysis

The statistical analysis was performed using EZR™ (version 1.55) software and the open‐source, cross‐platform software for advanced meta‐analysis ‘openMeta [Analyst]™’ version 12.11.14 by authors ERO and SHE [23]. The frequency of continuous variables was reported as mean ± standard deviation, and categorical variables as numbers and percentages (95% confidence interval [CI]). Meta‐analysis was performed by standard methodology using a random‐effect model. Statistical heterogeneity was assessed by inconsistency (I ²) statistics. The odds ratio of dichotomous outcomes was calculated along with its 95% CI.

RESULTS

Seventy‐two publications were screened, and three observational studies met the inclusion criteria. The three observational studies included one case–control (Level 3b), one cohort study (Level 4) and one survey (Level 5); selection criteria are shown in Figure 1. No randomized controlled trials were found. The included studies were conducted between 1979 and 2016 and comprised 27,061 adult patients who underwent IPAA. These surgeries were performed at multiple centres in two countries (Table 1).

FIGURE 1.

Selection criteria.

TABLE 1.

Profile of included studies.

Study	Year	Country	Study design	Study period	IPAA surgery patients	Salvage surgery population	Pouch prolapse population	Perineal salvage for pouch prolapses
Ehsan et al. [24]	2004	US	Survey	1979–2001	23,541	52	83	28
Joyce et al. [9]	2010	US	Cohort	NA	3,176	9	11	2
Rossi et al. [17]	2019	France	Case control	1999–2016	344	33	1	1
Total					27,061	94	95	31

Abbreviations: IPAA, ileoanal pouch anal anastomosis; NA, not available.

Prevalence

A total of 95 patients with pouch prolapse were reported in the three included studies. The prevalence of patients with pouch prolapse was 0.4% (95% CI 0.3–0.4; I ² = 0%) (Figure 2).

FIGURE 2.

Forest plot of the prevalence of ileal anal pouch prolapse.

Demographics

Demographic details were limited (Table 2). The age range of patients reported in two studies with a total of 27 patients was 31.6–38 years. Sex distribution was reported in only one study, comprising 11 patients with a male‐to‐female ratio of 1.2:1.

TABLE 2.

Demographics of patients with ileoanal pouch complications.

Study	Pouch prolapse population	Age (mean ± SD)	Male	Female	Body mass index (mean ± SD)
Joyce et al. [9]	11	31.6	6	5	NA
Rossi et al. a [17]	16	38 ± 11	8	8	22 ± 3
Ehsan et al. [24]	83	NA	NA	NA	NA

Abbreviations: NA, not available; SD, standard deviation.

^a General data available on all trans‐anal salvage surgeries for ileoanal pouch anal anastomosis complications, including a sole case of pouch prolapse (specific data for prolapse not available).

Clinical features

Time to prolapse from primary IPAA construction was heterogeneously reported as 2.6 years in one study and in another study as <2 years (48%), 3–5 years (29%), 6–8 years (5%) and >8 years (13%). Symptoms of pouch prolapse besides anal protrusion of tissue included straining (18/47; 38.3%), seepage (11/47; 23.4%) and anal pain (7/47; 14.9%) (Table 3). Full thickness prolapse was more frequently reported than mucosal prolapse in one study with details of prolapse category [7/11 (63.6%) vs. 4/11 (36.4%), respectively]. Indications for primary IPAA as documented in two studies were MUC (18; 66.7%), FAP (5; 18.5%), Crohn's disease (3; 11.1%) and dysmotility (1; 3.7%).

TABLE 3.

Presenting symptoms of pouch prolapse.

Study	Prolapse population	Prolapse (%)	Anal pain (%)	Dysfunction (%)	Seepage of stool (%)	Straining (%)	Fecal Incontinence (%)	Other (%)
Joyce et al. [9]	11	9 (81.8)	2 (1.8)	1 (0.9)	0 (0)	0 (0)	0 (0)	0 (0)
Rossi et al. [17]	1	1 (100)	0 (0)	0 (0)	1 (100)	0 (0)	0 (0)	0 (0)
Ehsan et al. [24]	83 a	22 (62.8)	5 (14.2)	0 (0)	10 (28.5)	18 (51.4)	6 (17.1)	8 (22.8)
Total	47	32	7	1	11	18	6	8

^a 35 respondents out of 83 surgeons surveyed.

Initial pouch surgery

Two studies with 21 (22.5%) cases reported a laparoscopic approach for initial pouch surgery, while the third study with 83 (77.5%) cases had no report on the surgical technique. J‐pouch was the predominant configuration in primary IPAAs in 9 of 12 cases, with initial pouch configuration reported (75%).

Salvage surgery

Generally, SPs for different IPAA complications were performed in 94 patients, with a pooled frequency of 0.5% (95% CI −0.0 to 1.1; I ² = 94.3%). Of 95 patients with IPAA prolapse, a perineal salvage approach was used in 31, with a pooled frequency of 26.2% (95% CI 11.0–63.5; I ² = 95.7%) (p < 0.001) (Figure 3).

FIGURE 3.

Forest plot of perineal salvage surgery for ileoanal pouch prolapse.

In the included studies, perineal SPs were less frequently performed for IPAA prolapse than an abdominal approach [OR 0.47 (95% CI 0.25–0.89; I ² = 52%)] (Figure 4). In terms of repair technique, pouch advancement was the most frequently reported [88.4% (95% CI 77.5–99.3)] (Table 4).

FIGURE 4.

Forest plot comparison of perineal and transabdominal approaches in salvage surgery for ileoanal pouch prolapse (abdominal salvage as control group).

TABLE 4.

Types of perineal salvage surgery for ileoanal pouch prolapse.

Study	Total number of perineal salvage surgeries	Pouch advancement surgery	Pouchpexy	Division of prolapsing septum
Joyce et al. [9]	2	2	0	0
Rossi et al. [17]	1	1	0	0
Ehsan et al. [24]	28	25	2 a	1
Total	31	28	2	1

^a Uncertain if pouchpexy was purely perineal SPs.

Outcomes

The overall morbidity rate was 23% from data available in two studies, and the overall mean success rates in the three studies were 75%, 96.4% and 100%, respectively (Table 5). The success rate of 100% was recorded in mucosal prolapse cases, while the other studies were not specific on the type of prolapse. There was a paucity of data on specific short‐ and long‐term outcomes, specifically recurrence. However, one of the studies with a cohort of 16 transanal SPs for pouch complications comprising a single case of prolapse reported a mean hospital stay of 6 days. A general report for all patients in this SPs cohort over a mean duration of 6 ± 4 years included a mean bowel movement/24 h of 7, a mean bowel movement/night of 1 (0–3) and urgency in one (8%) and incontinence in four (25%) patients. In addition, a mean Cleveland Global QoL score (CGQL) of 7 and a mean Cleveland Pouch Functional Score (PFS) of 4 was reported in this cohort. In terms of sexual function, the mean Female Sexual Function Index (FSFI) for females was 55 ± 24. The International Index of Erectile Function (IIEF‐5) for four male patients documented two cases each of erectile dysfunction and normal erectile function.

TABLE 5.

Outcome of perineal salvage surgery for ileoanal pouch prolapse.

Study	Hospital stays (days)	Overall morbidity (%)	Mean follow‐up duration (years)	Overall success rate (%)	Recurrence (%)
Joyce et al. [9]	NA	NA	5 a	100	0
Rossi et al. b [17]	6 ± 1	19	6 ± 4	75.0	NA
Ehsan et al. [24]	NA	27	NA	96.4	NA

Abbreviation: NA, not available.

^a Median value reported.

^b Data for 16 transanal salvage surgeries for IPAA.

Risk of bias

By consensus agreement, two studies were rated as having good quality of evidence, while the survey study was rated as poor quality of evidence (Table S1).

DISCUSSION

Salvage surgery is used for the management of complications of IPAA to preserve the pouch and maintain gut continuity. As mucosal and full thickness prolapse following IPAA is exceedingly rare, consequently, a treatment algorithm has not been well defined. In this comprehensive review of the literature over 37 years, the prevalence rates of IPAA prolapse ranged between 0.3% and 0.4% [9, 17, 24], with a pooled prevalence of 0.4%. Broadly, perineal SPs for pouch prolapse comprise pouch advancement with or without sphincter repair and pouchpexy with or without mesh placement. In this review, perineal SPs were less frequent than abdominal operations for repair of IPAA prolapse [OR 0.47 (95% CI 0.25–0.89; I ² = 52%)]. Rossi et al. [17] reported that 3% of prolapsed patients underwent a perineal approach versus 53.8% in the Ehsan et al. study [24] Different patient populations, indications and surgeon preferences may affect the uptake of this approach. Pouch advancement was the most common perineal procedure performed [88.4% (95% CI 77.5–99.3)], and an overall mean success rate of 90.5% over the follow‐up period was recorded. Interestingly, this result was higher than that reported for the success of trans‐perineal SPs for IPAA of 71.8%–77.3% [25, 26]. IPAA patients with septic complications have a higher chance of having a hand‐sewn anastomosis in the redo setting [27].

One suggested reason for the rare complication of prolapse is the traction placed on the pouch by the small bowel mesentery, thereby decreasing the opportunity for prolapse [9]. Other mechanical complications of IPAA include stenosis and stricture. However, the most common complication following IPAA is small bowel obstruction, with an incidence between 13% and 25%, which tends to increase with longer follow‐up [28]. The timing of prolapse from initial IPAA creation is highly variable. This has been attributed to multiple factors, from technical issues associated with pouch construction to patient loss to follow‐up [24]. Joyce et al. [9] reported a mean time to prolapse from index surgery of 2.6 years. In concordance with this, Ehsan et al. [24] reported the highest proportion of patients presented within 2 years. In terms of IPAA configuration, the J pouch was the most common. Generally, the common location of ileoanal prolapse is the anterior wall of the distal pouch body or cuff, with circumferential prolapse at the distal pouch, pouch inlet and dome at the tip of the J [29]. There was insufficient data to compare prolapse location and rates of the different pouch designs.

The indication for primary IPAA in our study population was predominantly MUC at 66.7%, followed by FAP at 18.5%, Crohn's disease at 11.1% and colonic dysmotility at 3.7%. An external protrusion of pouch tissue from the anus was by far the most common presentation at 81.8%, 100% and 62.8% in the three studies. This was followed by straining, seepage of stool and fecal incontinence. Other symptoms that are associated with pouch prolapse include a sense of incomplete evacuation, nausea, bloating, pruritus and perianal dermatitis [30]. Pouch prolapse shares a similar symptomatology profile to afferent efferent limb syndromes, enterocele, redundant loop and folding pouch, with an umbrella description as floppy pouch complex [8, 31]. A full patient assessment, including medical/surgical history, physical examination and investigations, should accurately diagnose the underlying condition. The relevant investigations include endoscopic, histologic, manometric and radiologic studies comprising plain abdominal radiographs, small‐bowel series, dynamic defecography, computerized tomography and magnetic resonance enterography. Full‐thickness prolapse was more commonly seen. In all, a decision on the choice of SPs for pouch prolapse is highly dependent on whether prolapse is mucosal or full thickness, with treatment tailored to anatomic abnormality.

If mucosal prolapse is confirmed, conservative measures including the use of stool bulking agents, biofeedback therapy, and endoscopic techniques of band ligation and snaring can be initiated [32]. In the event of non‐improvement of symptoms by initial conservative measures, surgical intervention is required. This category of pouch prolapse is amenable to a transanal advancement procedure with the excision of redundant mucosa. Full‐thickness pouch prolapse usually requires transabdominal surgery [19, 33, 34, 35]. Notably, many studies on abdominoperineal revisional and redo SPs for IPAA complications are collectively reported as abdominal approaches [10, 11, 28, 36]. The report of sole perineal SP for full thickness prolapse is limited in the literature and long‐term outcome results [37]. In this study, an overall morbidity rate of 23% was recorded for perineal SPs, which is lower compared to the transabdominal approach morbidity rate of 36%–61% [17, 18, 19]. Hence, it is advocated by authors that revisional surgery should first be attempted transanally in mucosal prolapse without fibrosis and in patients where an abdominal approach would be unwise, such as in frail patients with a high morbidity or history of mesh repair, with reduced morbidity (Figure 5).

FIGURE 5.

Algorithm for surgical management of prolapsed ileoanal pouch.

If surgical treatment is unsuccessful or is contraindicated, the only other available options are the creation of a new pouch or pouch excision and permanent ileostomy [38]. Revisional SPs involve abdominal exploration and correction of the pathology with or without abdomino‐anal advancement and with or without disconnection of the existing anastomosis and the creation of a new one [2]. In contradistinction, de novo creation of a pouch for the resection of an existing pouch is the scope of redo SPs [2]. Redo salvage surgery is feasible in highly motivated patients, but there is limited data on long‐term functional outcomes [16, 39]. One study reported complications being more frequent in patients who had pouch excision with de novo pouch creation compared to patients whose existing pouch was retained [25]. Thus, failure of one salvage attempt should not be considered a contraindication to additional attempt(s). Generally, IPAA patients following SPs have improved functional outcomes but a higher frequency of bowel movements than primary IPAA [40]. In addition, there is a higher reported rate of urgency and incontinence and lower CGQL scores in comparison to primary IPAA patients [26, 41]. As the volume of pouch prolapse surgeries increases, more prospective studies are needed on short‐ and long‐term functional outcomes of salvage and redo salvage perineal surgeries for pouch prolapse.

STUDY LIMITATIONS

No randomized controlled study on the research subject was identified, which decreased the level of evidence from our meta‐analysis. In addition, data were only from two countries and were predominantly obtained at high‐volume centres with large IPAA experiences. Hence, whether the technical success of perineal salvage surgery for pouch prolapse could be achieved in low‐volume centres requires further investigation. Furthermore, the included studies had some incompleteness related to baseline information (age, sex, and body mass index). Importantly, standardized criteria for patient selection into the two major perineal SPs (pouchpexy vs. pouch advancement) were not formulated, and differences among the studies relative to surgeon preference may have contributed to the heterogeneity in our pooled analysis. Meanwhile, our review had limited short‐ and long‐term outcomes to compare pouch advancement versus pouchpexy for pouch prolapse. Therefore, we could not conduct a subgroup analysis based on the type of perineal SPs that patients received. Although the trans‐anal pouch advancement was frequently reported and a high success rate was achieved, a subgroup analysis is needed to verify any potential relative advantages.

In conclusion, although perineal SPs were infrequently performed for the uncommon complication of IPAA prolapse, these procedures were associated with good outcomes, mainly in mucosal prolapse. These results may be useful in clinical practice and to guide further investigation in specialty IPAA centres.

AUTHOR CONTRIBUTIONS

Emeka Ray‐Offor: Conceptualization; investigation; writing – original draft; methodology; validation; visualization; formal analysis; data curation. Zoe Garoufalia: Investigation; data curation; writing – review and editing; formal analysis. Sameh Hany Emile: Investigation; formal analysis; data curation; writing – review and editing. Peter Rogers: Investigation; formal analysis; data curation; writing – review and editing. Nir Horesh: Investigation; formal analysis; data curation; writing – review and editing. Rachel Gefen: Investigation; formal analysis; data curation; writing – review and editing. Peige Zhou: Investigation; formal analysis; data curation; writing – review and editing. Giovanna DaSilva: Conceptualization; project administration; supervision; writing – review and editing. Victor Strassmann: Investigation; formal analysis; data curation; writing – review and editing. Steven D. Wexner: Conceptualization; project administration; supervision; writing – review and editing.

FUNDING INFORMATION

This research did not receive any specific grant from funding agencies in the public, commercial or not‐for‐profit sectors.

CONFLICT OF INTEREST STATEMENT

Dr. Wexner is a consultant for ActivSurgical, Arthrex, Baxter, Becton, Dickinson and Co., Intuitive Surgical, OstomyCure, Takeda, Virtual Ports, is chair of the Data Safety Monitoring Board of Polypoid and receives royalties from Intuitive Surgical, Karl Storz Endoscopy America Inc. and Unique Surgical Solutions, LLC. Dr. Emile is a consultant for Becton, Dickinson and Co. None of the other authors report any financial conflicts of interest.

Supporting information

Data S1:

Ray‐Offor E, Garoufalia Z, Emile SH, Rogers P, Horesh N, Gefen R, et al. Perineal salvage surgery for prolapse of ileal pouch anal anastomosis: Systematic review and meta‐analysis. Colorectal Dis. 2025;27:e70202. 10.1111/codi.70202

DATA AVAILABILITY STATEMENT

Upon reasonable request from first author.