Int J Colorectal Dis (2015) 30:1109–1115 DOI 10.1007/s00384-015-2223-9
ORIGINAL ARTICLE
Prophylactic surgery in familial adenomatous polyposis (FAP)—a single surgeon’s short- and long-term experience with hand-assisted proctocolectomy and smaller J-pouches Ralph Schneider 1 & Claudia Schneider 1 & Anne Dalchow 1 & Christian Jakobeit 2 & Gabriela Möslein 1
Accepted: 20 April 2015 / Published online: 3 May 2015 # Springer-Verlag Berlin Heidelberg 2015
Abstract Purpose Prophylactic proctocolectomy with an ileoanal neo-reservoir is the established procedure in nonattenuated familial adenomatous polyposis (FAP). Traditionally, the ileal J-pouch is created by doubling 15 cm of the terminal ileum. Pouch inlet problems are not infrequently encountered in longer pouches. On this rationale, this series reports on the functional outcome and quality of life (QoL) following standardized construction of a shorter J-pouch with a limb of 8–9 cm length. Methods All patients of a single-surgeon series with FAP who underwent hand-assisted laparoscopic proctocolectomy and small ileal pouch-anal anastomosis as the primary procedure between 10/2005 and 04/2010 and responded to the questionnaire were included and retrospectively analyzed. Results A total of 46 patients (78 %) out of the consecutive series who underwent operation in this period were included in the study. After a mean follow-up of 38 months, 40/46 patients (87 %) did not report any incontinence and 3 patients (6.5 %) complained about occasional nocturnal incontinence (3 failed to answer this question). The mean stool frequency per 24 h was 6.25. No significant difference was encountered between the QoL outcome of our patients versus the German normative population. Comparable results were achieved in a
* Ralph Schneider
[email protected] 1
Department of General and Visceral Surgery, Coloproctology, HELIOS St. Josefs-Hospital, Axstrasse 35, 44879 BochumLinden, Germany
2
Department of Gastroenterology, HELIOS St. Josefs-Hospital, Bochum-Linden, Germany
study analyzing the long-term results in FAP patients with a 15-cm pouch. Conclusions Smaller, 8–9 cm J-pouches show excellent functional results both in short- and in long-term results. The handassisted procedure was safe and no conversions were required. QoL is equal to a normative population, as it is in a series of patients with larger J-pouches. Keywords Familial adenomatous polyposis . Prophylactic surgery . Ileoanal pouch . Functional outcome . Quality of life
Introduction Familial adenomatous polyposis (FAP) is a rare autosomal dominant syndrome caused by a germline mutation in the adenomatous polyposis (APC) gene on chromosome 5 [1, 2]. The Bclassical^ syndrome is clinically characterized by hundreds to thousands of adenomas throughout the colorectum, inevitably leading to colorectal cancer in virtually all patients at an average age of 39 years [3, 4]. The attenuated variant of the syndrome is somewhat more benign with a later age of onset, sparser polyp burden, a more right-sided predominance of the neoplastic lesions, and frequently a sparing of the rectum [5]. Although some studies have demonstrated a beneficial effect of chemoprevention on colorectal neoplasia, the only curative prophylactic measure remains prophylactic surgery [6]. Colectomy with an ileorectal anastomosis (IRA) or a restorative proctocolectomy with an ileal pouch-anal anastomosis (IPAA) is the surgical procedure recommended for patients with FAP. Although colectomy with IRA has a good functional outcome regarding stool frequency, continence, and soiling, a high need of secondary proctocolectomies due to uncontrollable polyps [7–10] and a cumulative risk of cancer evolving
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in the rectal remnant of 15 % is reported after 25 years of follow-up [7, 11, 12]. Also, the high rate of desmoids in the mesentery following colorectal surgery impedes transformation of an IRA into an ileoanal pouch in more than 10 % of the patients [13]. Therefore, the recommended treatment for classical FAP with a high polyp burden in the rectum is restorative proctocolectomy with IPAA which has the advantage of removal of all or almost all of the colorectal mucosa and consecutively a minimal risk of rectal cancer [14–17]. However, this procedure is technically more demanding, has a higher risk of complications [3, 17], and especially the risk of loss of the small bowel due to secondary pouch excision caused by pouch failure [18, 19]. In addition, female fertility may be compromised [20, 21], and morbidities in the incidental need to construct a temporary diverting ileostomy including a second surgery for ileostomy closure triggering an increased risk of desmoid tumor formation at this site [22] must be considered in this equation. Moreover, less satisfactory functional results have been reported in short-term studies for IPAA compared to colectomy with IRA [8, 23–26]. Some studies correlate a poor functional outcome with proctomucosectomy and handsewn anastomosis [27–29]. Postoperative quality of life (QoL) is another important parameter for the evaluation of outcome for this complex surgical procedure. Taking into account the young age at time of surgery, this aspect gains higher importance in patients with classical FAP; additionally, life expectancy has increased with colorectal cancer being prevented in the majority of cases [3]. For quantification of QoL, both health-related and diseasespecific factors have been described and represent QoL as a function of several dimensions (e.g., physical function, social function, emotions, and symptoms). QoL nevertheless is difficult to assess, but standardized, validated questionnaires allow comparability of results [3]. However, in the context of ileoanal pouches, most studies analyze QoL in a mixed population of FAP and ulcerative colitis patients [30–32]. For comparability of our results, the report on outcome of FAP patients from the University of Heidelberg who underwent IPAA more than 10 years ago was selected as best reference. These authors describe IPAA as a safe surgical procedure with a high QoL comparable to that of a normative population [3]. Their long-term results are consistent with those reported in literature for shorter follow-up periods after IPAA [33–36]. Patients and methods All patients who underwent a laparoscopic hand-assisted proctocolectomy with a small ileoanal pouch in the Department of General and Visceral Surgery in the HELIOS St. Josefs-Hospital in Bochum-Linden/Germany between 10/2005 and 04/2010 were invited to complete a retrospective questionnaire. All participants provided written informed consent.
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The operation was performed by a single surgeon (G.M.), and all FAP patients in the time period treated by this standardized laparoscopic hand-assisted procedure were included in the study. A pouch was created using only one GIA 100 mm magazine, so the resulting length of the pouch was about 8 cm. A 0.5–2.0 cm residual rectal cuff of columnar epithelium was retained in the upper anal canal above the anal transitional zone. The rectal cuff was shortened as much as technically (anatomically) feasible. Due to the systematic double-staple technique using either the BTX30^ or the Bcontour stapler^ device, clearly the staple line was in the very low rectum. A protective loop ileostomy was optional. The decision to perform an ileostomy was made intraoperatively based on technical ease of anastomosis and mesenteric tension, primary impermeability of the anastomosis at control during the procedure, and patient’s preference. Known cancer at the time of surgery was an exclusion criterion for this study. The anastomoses were all performed in double-staple technique. In this series, no proctomucosectomy was performed, and in all cases, it was the primary prophylactic colorectal procedure for the patient. Indications for prophylactic surgery were based on size and distribution of polyps, increasing dysplasia in histology, and symptoms such as diarrhea and/or anemia. In all cases, the ileocolic arcade was preserved. All patients were invited to participate in the survey and to complete the German version of the Short Form 36 questionnaire (SF-36), a questionnaire to survey health-related QoL in any population [37] covering eight scales (physical functioning, role limitations due to physical problems, social functioning, bodily pain, general mental health, role limitations due to emotional problems, vitality, and general health). Additional questions regarding stool frequency in 24 h, continence during day/night/whole day, the Wexner score, the use of stool-regulating medication in addition to any regular medication, and diet were included. The QoL of the cohort was compared to that of a German normative population, which was assessed by the SF-36 [38, 39]. Data were analyzed using SPSS software (Statistical Product and Services Solutions, version 16.0, SPSS Inc, Chicago, IL, USA). All results are expressed as means±standard error of the mean and percentages. Fisher’s exact test was used to determine statistically significant differences among the groups. P values of less than 0.05 were considered to be statistically significant.
Results Out of 59 patients who underwent a laparoscopic handassisted prophylactic proctocolectomy and small ileoanal pouch in this period, 46 patients (78.0 %) were included in
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the study. Ten patients did not respond, two patients were lost to follow-up, and one patient still had a diverting ileostomy. The mean age at the time of the operation for the 46 patients included (21 males, 25 females) was 27.14±1.78 years. An abdominal wall or mesenteric desmoid developing during follow-up was confirmed via imaging (CT or MRI) in 11/46 patients (23.9 %) and suspected clinically in 2/46 patients (4.3 %). The length of follow-up is summarized in Fig. 1. After a mean follow-up of 38.1±3.0 months, 40/46 (87 %) described their sphincter function as entirely continent, 3 patients (6.5 %) reported nocturnal incontinence or soiling only, and 3 patients (6.5 %) did not answer the question. The mean stool frequency per day was 6.25±0.36 (median 5.5, range 3.5 to 12). A subgroup of five patients, who were less than 1 year after proctocolectomy, had a mean of five and a median of seven bowel movements. Thirty-two of forty-six patients (69.6 %) stated not to apply any medication for stool regulation or bulk-forming agents. Loperamide was intermittently used by 9/46 patients (19.6 %) and fibers such as psyllium were taken by 3/46 patients (6.5 %). Combined intake of both types of medication were reported by 2/46 patients (4.3 %). The results of the Wexner score show a mean score of 3.16 ±0.68, whereas a score of B0^ is completely continent and a score of B20^ is completely incontinent. The single mean scores are summarized in Table 1. As can be seen in Table 2, after performing a Bonferroni correction due to multiple testing, there is no significant difference between the QoL between our patients and a healthy normative population. The results are graphically displayed in Fig. 2. Almost all patients from this study were compliant with the follow-up recommendation of a yearly annual pouchoscopy in addition to a physical exam and ultrasound examination of the abdomen. Of the 40 patients that had their follow-up at our institution, relevant polyps in the rectal remnant requiring a mucosectomy were found in one patient with low-grade neoplasia. In two other patients, small adenomas were detected in the pouch without therapeutic intervention. These patients are attractive recruits for future chemoprevention studies. Fig. 1 Length of follow-up
1111 Table 1 Wexner score (0 points =continent; 20 points= complete incontinent). The mean scores±SEM for each Btype of incontinence^ is shown Type of incontinence
Patients with primary pouch (n=46)
Solid
0.66±0.18
Liquid
0.89±0.19
Gas Wear pad
0.80±0.18 0.31±0.14
Lifestyle altered Total
0.67±0.15 3.16±0.68
(0=never, 1=rarely, less than once per month, 2=sometimes, less than once a week, 3=usually, less than once a day, 4=always, every day)
Discussion In FAP patients with high polyp rectal polyp burden, restorative proctocolectomy and construction of an ileoanal pouch is the recommended procedure. The operation is technically demanding and has a substantial rate of complications. Nevertheless, a QoL similar to the general population can be achieved and functional results seem comparable to subtotal colectomy, though the functional outcome regarding the two procedures is still discussed controversially [6, 23, 40]. The functional results in this series of hand-assisted laparoscopic smaller pouches are excellent, and interestingly, the smaller reservoir does not lead to a higher frequency of bowel movements. These functional results with a mean daily stool frequency of 6.25±0.36 with no daytime incontinence and an incontinence rate of only 6.5 % during nighttime are actually comparable to functional results achieved with an IRA as published in a study by Ambroze [40]. The results are also better than the results of two other studies comparing IRA and IPAA [6, 23]. The mean stool frequency is comparable between our study and the IPAA cohort of Ambroze [40] and Madden [23], whereas the Dutch cohort [6] shows a higher frequency of bowel movements. The results are also comparable to a more recent study analyzing the functional outcome of 223 patients with FAP after IPAA, who reported an average of six bowel movements 1, 5, and 10 years postoperatively [41]. Therefore, no deterioration regarding the amount of bowel movements over time (10 years) was noted.
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Table 2 Mean scores of the SF-36 questionnaire in comparison to a healthy German normative population [39] Male
Female
Study
Control
Study
Control
PF
96,40
88,18
91,15
82,77
RP BP
88,25 83,08
85,53 71,04
82,33 80,04
79,22 63,89
GH
77,76
66,83
71,94
66,03
VT SF
69,00 91,50
62,58 88,63
67,45 84,00
57,57 84,24
RE MH
87,00 78,20
91,58 75,22
83,33 80,40
86,74 69,83
PF physical functioning, RP role limitations due to physical problems, BP bodily pain, GH general health, VT vitality, SF social functioning, RE role limitations due to emotional problems, MH mental health, higher score means better results p for all items was >0.5 after adjustment due to multiple testing
Initially, the correlation between the capacity of the pouch for collecting feces and functional outcome was determined [42, 43], and stool frequency has formerly been related to an inverse proportion with size and capacity of the pouch reservoir. Since then, the determination of the optimal length of the ileal limb used for pouch formation has been an issue of controversy. However, increasingly it has been realized that other factors in addition to the initial pouch volume determine functional short- and long-term outcomes [44]. The final volumetric capacity depends mainly on the length of the afferent loop [44].
An animal study on dogs using J- and S-pouches with afferent 6- or 2-cm loops and an initial 82–110 volume demonstrated that after 1 year J-pouches had doubled their reservoir capacity and S-pouches even tripled this volume [45]. The authors also demonstrated that pressure inside the pouch increases only if the myeloelectric activity involves both of the two or three branches of the pouch. Moreover, the contractile activity of the reservoir increased for height and frequency of the waves when the reservoir stretched. Considering these two facts, J-pouches seem to have a more effective motility and a better activity than the Spouches. Both J- and S-pouches showed a better function, when the length of the efferent loop was short [45]. Last but not least, the size of the pouch may have an important influence on the incidence of pouchitis, which occurs more frequently in patients with ulcerative colitis than in FAP after IPAA [44]. Assuming a better evacuation of smaller pouches with consecutive reduced fecal stasis, smaller pouches may be less susceptible to inflammation [44]. An important additional aspect in the technique of constructing an ileal pouch that has not been included in these studies is the aspect of maintaining circulation from the ileocolic vessels. At least in theory, maintaining blood circulation from both sides of the pouch with the ileocolic and mesenteric flow may improve healing of the anastomosis and better circulation may equally improve a disposition for inflammation leading to pouchitis. Especially for the more complication-prone group of ulcerative colitis patients, these potential benefits should be prospectively assessed. When comparing the results of QoL after proctocolectomy in different series, it is pivotal to differentiate between FAP and ulcerative colitis (UC) for several important reasons: &
&
& &
&
Fig. 2 Comparison between our patients and the German normative population
UC patients are ill at the time of prophylactic surgery and have experienced several years of disease and serious medication already. Frequently, UC patients are chronically ill at the time of proctocolectomy. UC patients requiring proctocolectomy are nonresponders to conservative therapy and as a rule have a chronically increased number of daily defecations and diarrhea. UC patients are more prone to perioperative complications such as fistulas [19, 46–48]. Incidence of pouchitis in UC patients has been reported to be as high as 50 % [49] compared to FAP patients with pouchitis in less than 11 % of cases [50]. UC patients do not develop desmoid tumors or upper GI polyposis.
Therefore, to the understanding of the authors, these patient groups cannot reasonably be compared in regard to functional pouch outcome or QoL. Ganschow et al. recently reported on a series of FAP patients with a functioning ileoanal pouch more than 10 years
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after primary surgery. QoL was assessed by the SF-36 and in their series, and FAP patients with IPAA had a comparable QOL to that of a German normative population [3]. The age at operation (28.7 versus 27.1 years in our study) is also comparable with our cohort, while the number of desmoids (17.5 versus 23.9 %) is lower in the Heidelberg group. This may be due to a variety of factors including the experience at the site of follow-up that may be non-institutional or to a selection of the patients included in their study. Also, the number of defecations (median 6 in the Heidelberg group versus 5.5 in our cohort) was comparable. Interestingly, both the long-term follow-up patients as reported by Ganschow et al. and our series with shorter follow-up showed an excellent QoL when compared with the normative population. The time of followup accounts for a limited comparability of both patient groups—on one hand because of the different age at the time of the survey and on the other because a change of pouch function and subsequently QoL may occur over time. In the study by Ganschow et al., the most important factor influencing patients’ QoL was the number of bowel movements. Fazio et al. reported on his series of 223 patients with FAP after IPAA. The median age at surgery was 31.9±12.4 years and the median follow-up 84 months (24–138 months). Of these patients, 188 received a J-pouch and 35 a S-pouch. The procedure was performed laparoscopically in only 30 patients. In this study, 81.6 % of the patients reported an incontinence Bnever/rarely^ 1 year postoperatively, 75.8 % 5 years postoperatively, and 73.6 % 10 years postoperatively. In comparison, in our study overall, 87 % of patients reported incontinence episode to be Bnever/rarely^ after an observation period of 38.1 months postoperatively, which is remarkably better than the best result of the above-reported study after the first year and overall best outcome year. Osterfeld in his publication reported that FAP patients initially experience a decrease in QoL, which however normalized in almost all parameters within the first postoperative year [51]. Therefore, our cutoff after 6 months postoperatively— and 5 (10.9 %) of our cohort were less than 1 year after proctocolectomy—also includes a group with an expected poorer outcome due to the shortened lapse of time between surgery and survey completion. Surprisingly, however, this group showed a mean of five and a median of seven bowel movements even at a shorter time after proctocolectomy or ileostomy closure. We conclude, therefore, that in our series, patients experience the benefit of a decreased frequency of bowel movements at an earlier postinterventional stage, despite the smaller pouch volume. As known from animal experiments, the volume of the J-pouch after 1 year is almost doubled when compared to the immediate postoperative volume [45], so further improvement of QoL in these patients may be postulated. The incidence of pouchitis in FAP patients varies widely in literature from about 5 % [52] to more than 20 % [53], and the
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definition of pouchitis is controversial. Furthermore, pouchitis is normally dealt with by general practitioners, so therefore data regarding the incidence of pouchitis in this series has not systematically been documented. For these reasons and the inconsistent definition of pouchitis, we did not analyze this parameter in our series. However, prospectively, this issue is of great relevance and should be systematically addressed in a larger cohort of patients on a prospective basis. Summarizing these facts, we have demonstrated that smaller pouches may be associated with better short- and long-term pouch evacuation and perform at the least equally when compared to a pure FAP cohort with a longer follow-up. Handassisted laparoscopic IPAA in our hands was readily feasible and did not require conversion in any of the consecutive cases. The aim of this study was to assess the functional short- and long-term functional results. Other theoretical advantages of smaller pouches that are hypothesized such as less inlet and outlet problems and less pouchitis are currently being prospectively documented in an evolving and growing series of primary proctocolectomies in FAP patients.
Conclusions Small J-pouches of 8–9 cm length with preservation of the ileocolic vessels were standardized in this series of FAP patients. The postoperative results measured with the validated SF-36 demonstrated excellent postoperative results in respect to both QoL and pouch function over the first 5 years. The capacity of the small pouch is sufficient to achieve very good function already in the early postoperative period. Further prospective studies regarding potential benefits of small pouches both regarding frequency and severity of pouchitis and pouch inlet morbidity for FAP in the long-term follow-up have been initiated. Also, the potential benefit of smaller pouches for UC patients remains to be determined.
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