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Original Report |
1 Department of Radiology, Hospital of the University of Pennsylvania, 3400
Spruce St., Philadelphia, PA 19104.
2 Department of Surgery, Hospital of the University of Pennsylvania,
Philadelphia, PA.
3 Department of Medicine, Hospital of the University of Pennsylvania,
Philadelphia, PA.
Received July 28, 2004;
accepted after revision September 15, 2004.
Address correspondence to M. S. Levine
(marc.levine{at}uphs.upenn.edu).
Abstract
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CONCLUSION. Pouch enemas showed characteristic findings of Crohn's disease, including nodularity, thickened folds, ulceration, cobblestoning, strictures, sinus tracks, and fistulas to the perianal region and vagina. It is important for radiologists to be aware of the findings of Crohn's disease in the ileal pouch and distal ileum on radiographic studies of the pouch after total proctocolectomy and ileal pouch-anal anastomosis for ulcerative colitis because of the implications for patient management.
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In previous studies, as many as 3.5-9% of all patients who underwent total proctocolectomy and ileal pouch-anal anastomosis for ulcerative colitis were found after surgery to have Crohn's disease, characterized by the development of severe pouchitis or by strictures, abscesses, sinus tracks, or fistulas involving the pouch or distal ileum [3-7]. Because of its intractable nature and associated complications, recurrent Crohn's disease in the ileal pouch often necessitates excision of the pouch with a permanent end ileostomy [3, 5, 7, 8], whereas nonspecific pouchitis after surgery for ulcerative colitis usually responds to medical treatment [9]. Because of the implications for patient management, it is important for radiologists to be aware of the findings on pouch enemas in patients with Crohn's disease involving the ileal pouch after surgery for ulcerative colitis. To our knowledge, the radiographic findings in this group of patients have not been reported previously in the radiologic literature. We therefore describe our experience with six patients who developed Crohn's disease in the ileal pouch after total proctocolectomy and ileal pouch-anal anastomosis for ulcerative colitis.
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Our institutional review board approved all aspects of this retrospective study and did not require informed consent from any patients whose records were included in our study.
Examination Technique
The pouch enemas were performed by injection of contrast material via a
22-French Foley catheter gently inserted into the ileal pouch. The studies
were obtained with a barium suspension (dilute Polibar Plus; E-Z-EM Company)
in four patients and a water-soluble contrast agent (Hypaque [diatrizoate
sodium]; Nycomed, or Gastroview [diatrizoate meglumine and diatrizoate
sodium]; Mallinckrodt) in two. Spot images and overhead radiographs of the
ileal pouch and distal ileum were obtained using digital fluoroscopy equipment
(Diagnost 76 Plus, Philips). All of the studies were performed by residents,
fellows, or one of three attending gastrointestinal radiologists, and all were
interpreted by the attending radiologists.
Image Review
The original radiologic reports and images for these six patients were
reviewed by a consensus of two of the authors (gastrointestinal radiologists
with 22 years and 20 years of experience, respectively) without knowledge of
the endoscopic or surgical findings (although the reviewers did know these
patients had a final diagnosis of Crohn's disease). The images were reviewed
to characterize the radiographic findings, including the presence or absence
of nodules, thickened folds, aphthoid ulcers, larger ulcers, cobblestoning,
sacculations, strictures, sinus tracks, fistulas, or other abnormalities
involving the pouch or distal ileum.
Study Design
Medical, endoscopic, and pathologic records also were reviewed by one
author to determine the clinical, endoscopic, and pathologic findings and the
treatment and subsequent course of these patients.
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Ileal abnormalities included narrowing and ulceration in two patients (Figs. 2 and 3), a cobblestone appearance with multiple linear and transverse ulcers in one (Fig. 1), strictures in three (two patients had one stricture and one had three strictures) (Fig. 3), and sacculations in one. In the five patients with concomitant ileal disease, the mean length of the diseased small bowel segment was 12 cm (range, 2-30 cm). Three patients had small bowel follow-throughs, and none of these studies showed more proximal small bowel involvement by Crohn's disease not visualized on the pouch enemas.
In all six cases, a diagnosis of Crohn's disease in the ileal pouch or in the pouch and distal ileum was suggested on the basis of the radiographic findings.
Treatment
Four patients received medical therapy with some combination of
antibiotics, steroids, immunosuppressive agents, and infliximab; two of these
four patients subsequently underwent excision of the ileal pouch with an end
ileostomy because of an inadequate clinical response to medical treatment. The
remaining two patients also underwent excision of the pouch and creation of an
end ileostomy without medical treatment. Thus, four (67%) of the six patients
required pouch excision with a permanent end ileostomy.
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Ulcerative colitis and Crohn's colitis usually have different clinical presentations and characteristic findings on radiologic, endoscopic, and pathologic examinations, enabling differentiation of these two forms of inflammatory bowel disease. However, overlap in the clinical, endoscopic, and histologic findings occurs in up to 15% of cases [9]. Even the classic histologic abnormality of Crohn's disease, the noncaseating granuloma, is found in only 50-60% of resected specimens [14]. Because of overlap in the diagnosis of ulcerative colitis and Crohn's disease, as many as 3.5-9% of patients who undergo total proctocolectomy and ileal pouch-anal anastomosis for definitive treatment of ulcerative colitis subsequently are found to have Crohn's disease in the ileal pouch [3-7], so that, in retrospect, these individuals presumably had surgery for Crohn's disease involving the colon rather than ulcerative colitis. In our series, the prevalence of Crohn's disease in the pouch was 5.8% (6/104), a figure within the range of earlier studies.
As in our series, patients with Crohn's disease in the ileal pouch may present with a variety of clinical findings, including abdominal pain, diarrhea, obstructive symptoms, draining perianal sinus tracks or cutaneous fistulas, and pouch-vaginal or pouch-vesical fistulas with passage of stool from the vagina or bladder [3-5, 7]. Although sinus tracks and fistulas occasionally may develop as complications of surgery, such findings should raise concern about the possibility of Crohn's disease involving the pouch (particularly if these sinus tracks and fistulas develop during the late postoperative period) in patients who undergo total proctocolectomy and ileal pouch-anal anastomosis for ulcerative colitis.
When patients with an ileal pouch-anal anastomosis develop clinical signs and symptoms related to the pouch, it is important to differentiate nonspecific pouchitis from recurrent Crohn's disease affecting the pouch. In patients originally treated for ulcerative colitis, nonspecific pouchitis is a common condition, with a cumulative 10-year incidence ranging from 24-46% [15-17]. Patients with pouchitis may present with symptoms of increased stool frequency; fecal urgency; abdominal and pelvic pain; and, less frequently, generalized malaise and fever [9], but they rarely develop strictures, abscesses, or fistulas. The onset of symptoms is variable, so the temporal relationship between ileal pouch-anal anastomosis and the development of pouchitis generally is not helpful for differentiating nonspecific pouchitis from Crohn's disease involving the pouch. Most patients with acute pouchitis have self-limited disease that responds to treatment with antibiotics, but some may develop chronic or relapsing pouchitis requiring maintenance medical therapy [9].
In contrast, Crohn's disease involving the ileal pouch or distal ileum tends to be a relapsing disease with a high morbidity related to complications such as strictures, fistulas, and abscesses [3-7]. As a result, these patients generally require aggressive medical management with some combination of steroids, immunosuppressive agents, and infliximab, the monoclonal chimeric antibody to tumor necrosis factor (TNF) [9, 18]. Although infliximab produces beneficial short-term or even long-term results in some cases [18], as many as 10-48% of patients with recurrent Crohn's disease in the ileal pouch have intractable disease, eventually necessitating excision of the pouch with a permanent end ileostomy [3, 5, 7, 8].
In our series of six patients with recurrent Crohn's disease after total proctocolectomy and ileal pouch--anal anastomosis, radiographic studies of the pouch revealed typical findings of Crohn's disease, including nodularity, thickened folds, ulceration, cobblestoning, strictures, sinus tracks, and fistulas to the perianal region and vagina (Figs. 1, 2, 3, 4). Five (83%) of the six patients also had findings of advanced Crohn's disease in the distal ileum (Figs. 1, 2, 3), but involvement of the more proximal small bowel was not seen (as is the case in most patients with ileal Crohn's disease). Although nonspecific pouchitis may also be manifested by nodularity, thickened folds, or even ulceration of the pouch, this condition ordinarily is not associated with cobblestoning, strictures, sinus tracks, or fistulas. Furthermore, patients with pouchitis would be very unlikely to have associated ileal disease [8]. We therefore believe that radiographic studies of the ileal pouch are extremely helpful for differentiating Crohn's disease affecting the pouch from nonspecific pouchitis, so that appropriate therapy can be instituted in these individuals.
It should be recognized that our series has the inherent limitations of a retrospective study. Our study design (i.e., investigating patients with Crohn's disease diagnosed on pouch enemas) also precluded determination of our radiographic sensitivity for detecting Crohn's disease in the ileal pouch in comparison to endoscopy. Furthermore, we only evaluated patients who were symptomatic because of advanced Crohn's disease involving the pouch and not asymptomatic patients with an ileal pouch. As a result, our prevalence (5.8%) of Crohn's disease in the ileal pouch may underestimate the true frequency of Crohn's disease in the pouch after total proctocolectomy and ileal pouch-anal anastomosis for ulcerative colitis. However, our series is intended primarily as a descriptive report to familiarize radiologists with the findings on pouch enemas in this group of patients.
In conclusion, we have reported six patients who underwent total proctocolectomy and ileal pouch-anal anastomosis for ulcerative colitis in whom radiographic studies of the pouch revealed typical findings of Crohn's disease in the ileal pouch or in the pouch and distal ileum. Such patients require intensive medical therapy and, not infrequently, excision of the pouch with a permanent end ileostomy. It is important for radiologists to be aware of the findings on pouch enemas in patients with Crohn's disease after ileal pouch-anal anastomosis because of the implications for patient management.
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