Spontaneous peritonitis from perforation of the colon in collagenous colitis

from Seattle, Washington, United States (4), a series of nine patients with collagenous colitis and colonic perforation were described. Although only limited details were provided, the investigators indicated that their patients with collagenous colitis developed perforations during or following colonoscopy or barium enema examinations, possibly related to impaired colonic wall compliance with air or barium insufflation. In the present report, no procedure was performed before the patients emergent presentation with an acute abdomen and peritonitis. It is conceivable that a second, independent cause for sigmoid colon ulceration could have been present, with the detection of collagenous colitis being only an unrelated and coincidental finding. Many causes for isolated colonic ulcers are listed in standard texts, but these could be readily excluded by this patients antecedent and limited clinical history, as well as the absence of any histological changes in her biopsies to suggest a second cause. In particular, Crohns disease of the colon was considered, because spontaneous Freeman et al Can J Gastroenterol Vol 15 No 4 April 2001 266 Figure 1) Colonoscopy in April 1997 showing normal-appearing colonic mucosa with the presence of a vascular pattern. Mucosal biopsies showed collagenous colitis Figure 2) Colonoscopy in April 1997 (same of Figure 1) showing an elliptical area of ulceration perforation of the colon may occur in this setting (7-9). Moreover, simultaneous collagenous colitis and Crohns disease have already been described (10). In this patient, however, there was no clinical or histological evidence for coexistent Crohns disease. Collagenous colitis has generally been considered a relatively benign disorder with chronic or episodic water diarrhea. It has few other significant complicating clinical disorders in the intestinal tract, except for celiac disease in the occasional patient (11); colon carcinoma complicating collagenous colitis has been described in a single patient (12). Although it has been suggested that instrumentinduced perforation may occur in this entity (4), the present report indicates that more serious intestinal complications may develop spontaneously in collagenous colitis. Perforation in collagenous colitis Can J Gastroenterol Vol 15 No 4 April 2001 267 REFERENCES 1. Freeman HJ, Weinstein WM, Shnitka TK, Wensel RH, Sartor VE. Watery diarrhea syndrome associated with a lesion of the colonic basement membrane-lamina propria interface. Ann R Coll Phys Surg Can 1976;9:45. 2. Lindstrom CG. Collagenous colitis with watery diarrhea  a new entity? Pathol Eur 1976;11:87-9. 3. Freeman HJ. Collagenous inflammatory mucosal diseases of the gastrointestinal tract. Can J Gastroenterol 1990;4:196-200. 4. Taylor SL, Haggitt RC, Bronner MP. Colonic perforation complicating colonoscopy in collagenous colitis (G4082). Gastroenterology 1999;116:A938. (Abst) 5. Bohr J, Tysk C, Eriksson S, Abrahamsson H, Jarnerot G. Collagenous colitis: a retrospective study of clinical presentation and treatment in 163 patients. Gut 1996;39:846-51. 6. Mullhaupt B, Guller U, Anabitarte M, Guller R, Fried M. Lymphocytic colitis: clinical presentation and long term course. Gut 1998;43:629-33. 7. Fisher J, Mantz F, Calkins WG. Colonic perforation in Crohns disease. Gastroenterology 1976;71;835-8. 8. Suk CH, Posner GL, Bopaiah V. Colonic perforation in Crohns disease. Am J Gastroenterol 1984;79:695-6. 9. Steinberg DM, Cooke WT, Alexander-Williams J. Free perforation in Crohns disease. Gut 1973;14:187-90. 10. Chandratre S, Bramble MG, Cooke WM, Jones RA. Simultaneous occurrence of collagenous colitis and Crohns disease. Digestion 1987;36:55-60. 11. Hamilton I, Sanders S, Hopwood D, Bouchier IA. Collagenous colitis associated with small intestinal villous atrophy. Gut 1986;27:1394-8. 12. Gardener GW, Goldberg R, Currie D, Murray D. Colonic carcinoma associated with an abnormal collagen table. Cancer 1984;54:2973-7.


CASE PRESENTATION
A 37-year-old woman presented with an acute abdomen in March 1997.This was preceded for three weeks by watery, nonbloody diarrhea.Laparoscopy demonstrated peritonitis, but no source could be defined; therefore, an exploratory laparotomy was done.Definite thickening and an inflammatory change in the sigmoid colon were detected, but there was no diverticular disease.No small bowel or mesenteric abnormalities were identified.Free intraperitoneal purulent material was present.A single lymph node in the sigmoid mesentery was removed; pathological evaluation showed only reactive changes.The patient was given intravenous cefoxitin.After recovery from surgery and discharge from hospital, she continued to have watery diarrhea.Colonoscopy revealed normal-appearing colonic mucosa (Figure 1) except for a focal elliptical ulcer in the sigmoid colon (Figure 2).Mucosal biopsies from different sites in the colon showed collagenous colitis.No granulomatous inflammatory change was seen.She was subsequently treated with 5-aminosalicylate, and her intake of wheat, gluten, milk and other dairy products was restricted until her diarrhea was resolved.A small intestinal biopsy was offered to the patient while she was on a normal diet to exclude occult celiac disease, but she refused.No further diarrhea or other gastrointestinal symptoms have been reported, and she remains well to date (October 1999).

DISCUSSION
This patient presented with an acute abdomen and peritonitis, apparently related to spontaneous sigmoid perforation.Postoperative studies also revealed histological changes of collagenous colitis, which account for her watery diarrhea.Although other studies, particularly a small intestinal biopsy, could not be done because the patient declined further evaluation, her presentation with spontaneous peritonitis was both dramatic and, apparently, unique.There are no prior case reports of this complication in either collagenous colitis or another form of microscopic colitis known as lymphocytic colitis.In addition, large series of patients with collagenous colitis (5) failed to identify this serious and life-threatening complication.Sudden onset of watery diarrhea and abdominal pain were frequent presenting symptoms in a series of 163 patients with collagenous colitis from 25 Swedish hospitals ( 5), but none had an intestinal perforation.Moreover, long term studies of large numbers of patients with lymphocytic colitis have also failed to record this complication (6).Interestingly, however, in a recent abstract from Seattle, Washington, United States (4), a series of nine patients with collagenous colitis and colonic perforation were described.Although only limited details were provided, the investigators indicated that their patients with collagenous colitis developed perforations during or following colonoscopy or barium enema examinations, possibly related to impaired colonic wall compliance with air or barium insufflation.In the present report, no procedure was performed before the patient's emergent presentation with an acute abdomen and peritonitis.
It is conceivable that a second, independent cause for sigmoid colon ulceration could have been present, with the detection of collagenous colitis being only an unrelated and coincidental finding.Many causes for isolated colonic ulcers are listed in standard texts, but these could be readily excluded by this patient's antecedent and limited clinical history, as well as the absence of any histological changes in her biopsies to suggest a second cause.In particular, Crohn's disease of the colon was considered, because spontaneous  1) showing an elliptical area of ulceration perforation of the colon may occur in this setting (7)(8)(9).Moreover, simultaneous collagenous colitis and Crohn's disease have already been described (10).In this patient, however, there was no clinical or histological evidence for coexistent Crohn's disease.
Collagenous colitis has generally been considered a relatively 'benign' disorder with chronic or episodic water diarrhea.It has few other significant complicating clinical disorders in the intestinal tract, except for celiac disease in the occasional patient (11); colon carcinoma complicating collagenous colitis has been described in a single patient (12).Although it has been suggested that instrumentinduced perforation may occur in this entity (4), the present report indicates that more serious intestinal complications may develop spontaneously in collagenous colitis.

Figure 1 )Figure 2 )
Figure 1) Colonoscopy in April 1997 showing normal-appearing colonic mucosa with the presence of a vascular pattern.Mucosal biopsies showed collagenous colitis