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Conundrum of the Cocoon Report of a Case and Review of the Literature Ambil S. Rajagopal, F.R.C.S (Edinb.), Ramasamy Rajagopal, M.S. From the Department of Surgery, PSG Institute of Medical Sciences and Research, Coimbatore, India Idiopathic, sclerosing, encapsulating peritonitis, or abdominal cocoon, is a rare cause of bowel obstruction in the elderly. A 65-year-old male patient presented with acute bowel obstruction caused by torsion of the encapsulated small bowel. He was treated successfully with subtotal excision of the fibrocollagenous membrane and small bowel resection. Despite anecdotal reports of a preoperative diagnosis, in the majority of cases, sclerosing, encapsulating peritonitis is a fortuitous finding. A better awareness of this condition may facilitate preoperative diagnosis. [Key words: Sclerosing peritonitis] Rajagopal AS, Rajagopal R. Conundrum of the cocoon: report of a case and review of the literature. Dis Colon Rectum 2003;46:1141–1143. I diopathic, sclerosing, encapsulating peritonitis (SEP), or “abdominal cocoon” as it is descriptively called, is a rare and enigmatic condition of unknown etiology. First described in 1978 in young girls, it was thought to be caused by a low-grade peritonitis from retrograde menstruation.1,2 Later, there were sporadic reports in older patients and in both genders.3 In this case, the small bowel becomes encased in a thick membrane not unlike a cocoon. It usually presents as an acute small bowel obstruction, as in this patient. REPORT OF A CASE A 65-year-old, apparently healthy patient presented with a history of diffuse abdominal pain, vomiting, and constipation of four days duration. There was no history of fever, recent weight loss, or appetite loss. His bladder habits were normal. There was no significant past medical history, history of abdominal surgery, or liver disease. He was not taking any medication. On physical examination, he was afebrile, the pulse rate was 90 per min, and blood pressure was 140 per 90 mmHg. He was not jaundiced, and t; here was no pallor. His abdominal examination revealed a flat abdomen with diffuse lower abdominal guarding and tenderness. There was no palpable abdominal mass, Address reprint requests to Mr. Rajagopal: Associate Professor of Surgery, PSG Institute of Medical Sciences and Research, Coimbatore, India. and bowel sounds were absent. Rectal examination was normal. He also had a partially reducible nontender left inguinal hernia. Blood investigation revealed a total white cell count of 13,000 and polymorphs 80 percent; other biochemical parameters and liver function test were normal. A plain x-ray of the abdomen showed few dilated small bowel loops with fluid levels. An ultrasound scan of the abdomen revealed dilated small bowel loops with minimum free fluid in the abdomen. Emergency laparotomy was performed for the acute small bowel obstruction. Exploration revealed encapsulation of the entire small bowel in a thick, white, adherent membrane (Figs. 1A and B). This was in two distinct masses: a proximal one containing the jejunum, and the distal one containing the ileum with torsion. The proximal encapsulation could be released, but we were unable to release the distal encapsulation, and this attempt resulted in a perforation. There also was a patch of gangrene over the distal ileum. Therefore a resection of the encapsulated ileum and end-to-end anastomosis of the small bowel was performed. He had an uneventful postoperative period. The peritoneal fluid was sterile on culture. Morbid anatomic and histopathologic examination were consistent with peritoneal encapsulation with a fibrocollagenous membrane, sclerosing encapsulating peritonitis (abdominal cocoon). DISCUSSION In the literature, abdominal cocoon is a mixed bag of both congenital and acquired conditions. For the sake of clarity, we should separate the congenital “peritoneal encapsulation,” an embryological abnormality, from sclerosing encapsulating peritonitis. In congenital encapsulation, the accessory peritoneal sheath, which is adherent to the bowel, is derived from the yolk sac lining as the physiologic umbilical hernia reduces into the general coelomic cavity during the twelfth week of gestation.4 The idiopathic SEP should be differentiated from that secondary to other conditions. Common among them are chronic perito- 1141 1142 RAJAGOPAL AND RAJAGOPAL Dis Colon Rectum, August 2003 Figure 1. A. Resected, encapsulated small bowel with partially excised, thick, adherent membrane. B. Concertina arrangement of bowel inside the cocoon on cross-section. neal dialysis, peritoneovenous shunting, and beta blocker treatment.5–7 Rarely it also is associated with abdominal tuberculosis, sarcoidosis, familial Mediterranean fever, gastrointestinal malignancy, protein-S deficiency, after liver transplantation, fibrogenic foreign material, and luteinized ovarian thecomas. The incidence of SEP in chronic peritoneal dialysis is 0.54 to 7.3 percent.8 An idiopathic fibrosclerotic disorder, such as retroperitoneal fibrosis, is probably unrelated to SEP.9 To confuse the issue, even a congenital theory for idiopathic SEP has been proposed, a chronic inflammatory process of “developmental peritoneal encapsulation,” which looks like a cocoon.10 In congenital peritoneal encapsulation, the membrane usually can be stripped easily, because it is predominantly mesothelial and not fibrocollagenous as in SEP. Contrary to original reports, SEP is neither peculiar to the tropics or subtropics nor is it exclusive to adolescent girls. It has been described in all age groups and both genders, as in this elderly, male patient. Amazingly SEP is not common in patients with repeated adhesive bowel obstructions or in cases of septic peritonitis. It usually presents as bowel obstruction, either caused by torsion of bowel, as in this case, or extraluminal compression by a constricting band. It also can present as chronic abdominal pain with malnutrition and as a vague abdominal mass. There are occasional instances of a preoperative diagnosis of SEP, either with a barium follow-through or after a contrast CT scan. The various imaging findings include peritoneal and bowel calcification, bowel wall thickening, loculated ascites, circumscribed mass of bowel loops conglomerated in one area (“cauliflower sign”), and delayed bowel transit.11–13 Essentially the diagnosis of SEP in the majority of cases is postexploratory, because it is a very rare cause of “surgical abdomen” and is not anticipated, as in this patient. A plain x-ray of the abdomen only indicates multiple fluid levels like in any other case of small bowel obstruction. Morphologically the cocoon is a thick, white membrane of fibrocollagenous nature and adherent to the bowel wall with interloop adhesions. The membrane characteristically encases the bowel in a concertina fashion as seen in the cross-section in Figure 1B. At times it is impossible to peel it off or excise it without perforating the bowel, as in this case. Various treatment options have been adopted in the past, such as subtotal excision of the membrane, enterolysis with partial excision, bowel resection, and exploratory laparotomy only. A bowel resection is not commonly needed if the membrane can be stripped atraumati- Vol. 46, No. 8 SCLEROSING, ENCAPSULATING PERITONITIS cally and obstruction released. Mortality of an anastomotic leak after bowel resection is significantly high, and in fact, some studies advice a proximal stoma in difficult cases of SEP.14,15 Recently there have been isolated reports of successful conservative management with corticosteroids, immunosuppressive therapy, and tamoxifen in SEP secondary to chronic peritoneal dialysis, in which it is a known complication.16–18 CONCLUSION Idiopathic, sclerosing, encapsulating peritonitis is a poorly understood and rarely documented cause of acute bowel obstruction. Although commonly reported in young girls, it also is seen in elderly, male patients. It is not unique to the tropics, and it is essentially a postexploratory finding, because it is not anticipated. REFERENCES 1. Foo KT, Ng KC, Rauff A, Foong WC, Sinniah R. Unusual small intestinal obstruction in adolescent girls: the abdominal cocoon. Br J Surg 1978;65:427–30. 2. Marinho A, Adelusi B. The abdominal cocoon: a case report. Br J Obstet Gynaecol 1980;87:249 –50. 3. Lifschitz O, Tiu J, Sumeruk RA. Peritoneal encapsulation of small intestine. A case report. S Afr Med J 1987;71: 452. 4. Sayfan J, Adam YG, Reif R. Peritoneal encapsulation in childhood. Case report, embryologic analysis and review of literature. Am J Surg 1979;138:725–7. 5. Nolph KD, Lindblad AS, Novak JW. Continuous ambulatory peritoneal dialysis. N Engl J Med 1988;318:1595– 600. 6. Stanley MM, Reyes CV, Greenlee HB, et al. Peritoneal fibrosis in cirrhotics treated with peritoneo-venous shunting for ascites. An autopsy study with clinical correlations. Dig Dis Sci 1996;41:571–7. 7. Grefberg N, Nilsson P, Andreen T. Sclerosing obstructive peritonitis, beta-blockers, and continuous ambulatory peritoneal dialysis. Lancet 1983;2:733– 4. 1143 8. Jenkins SB, Lang BL, Shortland JR, Brown PW, Wilkie ME. Sclerosing encapsulating peritonitis: a case series from a single U. K. center during a 10-year period. Adv Perit Dial 2001;17:191–5. 9. Dehner LP, Coffin CM. Idiopathic fibrosclerotic disorders and other inflammatory pseudotumors. Semin Diagn Pathol 1998;15:161–73. 10. Tsunoda T, Mochinaga N, Eto T, et al. Sclerosing encapsulating peritonitis combined with peritoneal encapsulation. Arch Surg 1993;128:353–5. 11. Krestin JP, Kacl G, Hauser M, et al. Imaging diagnosis of sclerosing peritonitis and relation of radiologic signs to the extent of the disease. Abdom Imaging 1995;20: 414 –20. 12. Stafford Johnson DB, Wilson TE, Francis IR, Swatz R. CT appearance of sclerosing peritonitis in patients on chronic ambulatory peritoneal dialysis. J Comput Assist Tomogr 1998;22:295–9. 13. Navani S, Shah P, Pandya S, Doctor N. Abdominal cocoon: “the cauliflower sign” on barium small bowel series. Indian J Gastroenterol 1995;14:19. 14. Kittur DS, Korpe SW, Raytch RE, Smith GW. Surgical aspects of sclerosing encapsulating peritonitis. Arch Surg 1990;125:1626 – 8. 15. Celicout B, Levard H, Hay J, Msika S, Fingerhut A, Pelissier E. Sclerosing encapsulating peritonitis: early and late results of surgical management in 32 cases. French Association for Surgical Research. Dig Surg 1988;15:697–702. 16. Mori Y, Matsuo S, Sutoh H, et al. A case of a dialysis patient with sclerosing peritonitis successfully treated with corticosteroid therapy alone. Am J Kidney Dis 1997;30:275– 8. 17. Martins LS, Rodrigues AS, Cabrita AN, Guimaraes S. Sclerosing encapsulating peritonitis: a case successfully treated with immunosuppression. Perit Dial Int 1999;19: 478 – 81. 18. Allaria PM, Giangrande A, Gandini E, Pisoni B. Continuous ambulatory peritoneal dialysis and sclerosing encapsulating peritonitis: tamoxifen as a new therapeutic agent. J Nephrol 1999;12:395–7.