اصلی Diseases of the Colon & Rectum Conundrum of the Cocoon

Conundrum of the Cocoon

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جلد:
46
ژبه:
english
صفحه:
3
DOI:
10.1007/s10350-004-7295-5
Date:
August, 2003
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PDF, 429 KB
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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-

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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.

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