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Case Reports and Images in Surgery

 
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Case Report
 
Abdominal cocoon: Intestinal obstruction, perforation and ischemia
Bassem Mahmoud Abou Hussein1, Ali Mohammad Al-Ani2, Filza Khalid Khalid3, Yousif Hussein El Tayyeb4, Alya Saif Al-Mazrouei4, Faisal Mohammad Badri4
1MD, Specialist, General Surgery Department, Rashid Hospital-Dubai Health Authority, Dubai, United Arab Emirates.
2MD, Senior Specialist, General Surgery Department, Rashid Hospital, Dubai Health Authority, Dubai United Arab Emirates.
3MD, Resident, General Surgery Department, Rashid Hospital-Dubai Health Authority, Dubai, United Arab Emirates.
4MD, Consultant, General Surgery Department, Rashid Hospital-Dubai Health Authority, Dubai, United Arab Emirates.

Article ID: 100021Z12BH2016
doi:10.5348/Z12-2016-21-CR-13

Address correspondence to:
Bassem Mahmoud Abou Hussein
United Arab Emirates
Dubai
Oud Meitha Street, Rashid Hospital, General Surgery Department, PO Box: 4545

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How to cite this article:
Abou Hussein BM, Al-Ani A M, Khalid F, El Tayyeb YH, Al-Mazrouei AS, Badri FM. Abdominal cocoon: Intestinal obstruction, perforation and ischemia. J Case Rep Images Surg 2016;2:46–49.


Abstract
Introduction: Abdominal cocoon, also known as sclerosing encapsulating peritonitis, is a rare condition that indicates total or partial encapsulation of the small bowel by a fibrocollagenous membrane or cocoon leading to bowel obstruction. Approximately 50 cases have been reported in literature. Patients usually present with a picture of bowel obstruction and most cases are diagnosed during laparotomy.
Case Report: We report a case of a 31-year-old male who presented with picture of bowel obstruction and a palpable para-umbilical abdominal swelling. Laboratory and radiologic investigations were not conclusive. Exploratory laparotomy showed that the patient has abdominal cocoon with bowel obstruction, perforation and ischemia. The capsule was removed and the involved bowel segment was resected.
Conclusion: Abdominal cocoon syndrome should be suspected in patients with bowel obstruction with no clear cause. Early diagnosis and treatment avoids bowel ischemia or further complications.

Keywords: Abdominal cocoon, Intestine, Ischemia, Obstruction, Perforation


Introduction

Abdominal cocoon, also known as sclerosing encapsulating peritonitis, is a rare condition that indicates total or partial encapsulation of the small bowel by a fibrocollagenous membrane or cocoon leading to bowel obstruction [1]. It is usually classified as primary (idiopathic) or secondary type. The primary type mainly presents in adolescent females from tropical countries and peritonitis evoked by retrograde menstruation may be a causative factor in this form. The second type is more common and is usually considered to be multifactorial, but genetic susceptibility may also be an etiological agent [2].

Clinically, most patients present with symptoms and signs suggestive of acute or bowel obstruction [3]. Management usually needs excision of the peritoneal sac and release of adhesions [4].


Case Report

A 31-year-old male patient, known to have alcoholic liver disease, portal hypertension and esophageal varices; presented to the emergency department complaining of central abdominal pain of one day duration. Pain was colicky, moderate in intensity, non-radiating, increased after food intake and associated with one episode of bilious vomiting. He reported a similar attack one week prior to presentation, which resolved spontaneously. He also gave history of a swelling in the left para umbilical area for one day, which also appeared two weeks before that, but disappeared spontaneously.

On examination: The patient was conscious, oriented and in pain. Vital signs showed pulse 70 bpm, blood pressure 104/66 mmHg and temperature 37°C. The abdomen was mildly distended but lax with a 3x3 cm soft, compressible, lobulated swelling in the left paraumbilical region, slightly tender, mobile, and non-pulsatile. No guarding or rigidity was present and bowel sounds were positive.

Laboratory investigations showed white blood cell 7000/ mm3, hemoglobin 7.4 g/dl, total bilirubin 1.5 mg/dl, creatinine: 3.5 g/dl, lactic acid 1.0, and C-reactive protein 39.

Computed tomography scan of the abdomen and pelvis showed cirrhotic liver changes associated with significant abdominopelvic ascites. Loops of small bowel were amalgamated in the left paraumbilical region with surrounding capsule (Figure 1) and (Figure 2).

Management
The patient was admitted to the hospital, started on intravenous fluids and antibiotics, and was shifted to the operating room.

Exploratory laparotomy showed:

  • Two liters of turbid, foul-smelling fluid in the abdomen.
  • Amalgamated small bowel mass encased in fibrous peritoneal pockets of fibrosis with areas of necrosis and perforation, (Figure 3).
  • Features were representative of abdominal cocoon with bowel perforation (Figure 4).
  • Parietal peritoneum showed extensive fibrous formation encasing the liver, colon and stomach.

Resection of the perforated small bowel segment was done.

The patient was transferred to the surgical intensive care unit (SICU) postoperatively and a relook laparotomy was done after one day.

Some of the fibrous tissue encasing the small intestine was released and an end-to-end anastomosis was created with a healthy bowel segment, but the patient was going into sepsis.

On pathologic examination, resected small bowel showed features of subacute serositis, while the capsule removed showed connective fibrous tissue.

The patient was then kept in the SICU; he developed an enterocutaneous fistula. Few days later, the patient started to have sepsis that resulted in multi-organ failure and death.


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Figure 1: Computed tomography of the abdomen and pelvis showing cirrhotic liver changes associated with significant abdominopelvic ascites with amalgamated loops of small bowel in the left paraumbilical region with surrounding capsule.



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Figure 2: Computed tomography of the abdomen and pelvis showing cirrhotic liver changes associated with significant abdominopelvic ascites with amalgamated loops of small bowel in the left paraumbilical region with surrounding capsule.



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Figure 3: Exploratory laparotomy showing amalgamated small bowel mass encased in fibrous peritoneal pockets of fibrosis with areas of necrosis and perforation.



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Figure 4: Exploratory laparotomy showing features representative of abdominal cocoon with bowel perforation.



Discussion

Abdominal cocoon or sclerosing encapsulating peritonitis (SEP) is a rare condition that refers to total or partial encapsulation of the small bowel by a fibrocollagenous membrane or cocoon with local inflammatory infiltrate leading to acute or chronic bowel obstruction [1]. The abdominal cocoon was first described and named in 1978 by Foo et al. Since then, approximately 50 cases have been reported in literature [5]. The majority of the cases are reported from tropical and subtropical climate belts of the world. The only case reported from a non-tropical zone is from England, in which the case was born in Pakistan [6].

The SEP is mainly an acquired disease. Many causes were listed in the pathogenesis of SEP including previous abdominal surgery or peritonitis, chronic ambulatory peritoneal dialysis and prolonged use of practolol [3]. Other conditions have been mentioned including ventriculoperitoneal and peritoneovenous shunts, SLE, sarcoidosis, cirrhosis, propranolol use for constrictive pericarditis, uterine and ovarian tumors, and recurrent peritonitis [7][8][9].

Clinically, most patients with abdominal cocoon syndrome (ACS) present with features of recurrent acute or chronic small bowel obstruction secondary to kinking and/or compression of the intestines within the constricting cocoon [3]. Yip and Lee [9] listed four main clinical features that help to identify abdominal cocoon preoperatively including: young girl with no other cause of bowel obstruction, similar attacks that resolved spontaneously, abdominal pain and vomiting, and a non-tender soft abdominal mass on palpation.

Abdominal X-ray and ultrasonography are not useful to differentiate ACS from other causes of bowel obstruction. Computed topography is more beneficial in differential diagnosis. The finding of a fibrous membrane enclosing the intestinal loops is a typical radiological finding of ACS [10]. However, although radiological findings, especially computed tomography, are helpful; yet, final and definite diagnosis is generally by surgery, as confirmed by ACS literature [10].

Treatment consists of excision of the peritoneal sac with lysis of the interloop adhesions. Bowel resection is done if a nonviable segment is found [4].


Conclusion

Abdominal cocoon is a rare cause of intestinal obstruction. It should be suspected in all patients with bowel obstruction of unknown cause. A high index of suspicion is needed for diagnosis. Treatment consists of adhesiolysis and excision of the cocoon. Early diagnosis and treatment may help to avoid bowel ischemia or perforation.


References
  1. Kirshtein B, Mizrahi S, Sinelnikov I, Lantsberg L. Abdominal cocoon as a rare cause of small bowel obstruction in an elderly man: report of a case and review of the literature. Indian J Surg 2011 Jan;73(1):73–5.   [CrossRef]   [Pubmed]    Back to citation no. 1
  2. Oran E, Seyit H, Besleyici C, Ünsal A, Alis H. Encapsulating peritoneal sclerosis as a late complication of peritoneal dialysis. Ann Med Surg (Lond) 2015 Apr 2;4(3):205–7.   [CrossRef]   [Pubmed]    Back to citation no. 2
  3. Cambria RP, Shamberger RC. Small bowel obstruction caused by the abdominal cocoon syndrome: possible association with the LeVeen shunt. Surgery 1984 Apr;95(4):501–3.   [Pubmed]    Back to citation no. 3
  4. Al-Abassi AA, Emad M. Abdominal cocoon. An unusual cause of intestinal obstruction. Saudi Med J 2004 Oct;25(10):1482–5.   [Pubmed]    Back to citation no. 4
  5. Hur J, Kim KW, Park MS, Yu JS. Abdominal cocoon: preoperative diagnostic clues from radiologic imaging with pathologic correlation. AJR Am J Roentgenol 2004 Mar;182(3):639–41.   [CrossRef]   [Pubmed]    Back to citation no. 5
  6. Yeniay L, Karaca CA, Caliskan C, Firat O, Ersin SM, Akgün E. Abdominal cocoon syndrome as a rare cause of mechanical bowel obstruction: report of two cases. Ulus Travma Acil Cerrahi Derg 2011 Nov;17(6):557–60.   [CrossRef]   [Pubmed]    Back to citation no. 6
  7. Krestin GP, Kacl G, Hauser M, Keusch G, Burger HR, Hoffmann R. Imaging diagnosis of sclerosing peritonitis and relation of radiologic signs to the extent of the disease. Abdom Imaging 1995 Sep-Oct;20(5):414–20.   [CrossRef]   [Pubmed]    Back to citation no. 7
  8. Kaushik R, Punia RP, Mohan H, Attri AK. Tuberculous abdominal cocoon--a report of 6 cases and review of the Literature. World J Emerg Surg 2006 Jun 27;1:18.   [CrossRef]   [Pubmed]    Back to citation no. 8
  9. Yip FW, Lee SH. The abdominal cocoon. Aust N Z J Surg 1992 Aug;62(8):638–42.   [CrossRef]   [Pubmed]    Back to citation no. 9
  10. Gupta S, Shirahatti RG, Anand J. CT findings of an abdominal cocoon. AJR Am J Roentgenol 2004 Dec;183(6):1658–60.   [CrossRef]   [Pubmed]    Back to citation no. 10

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Author Contributions
Bassem Mahmoud Abou Hussein – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Ali Mohammad Al-Ani – Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Filza Khalid Khalid – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Critical revision of the article
Yousif Hussein El Tayyeb – Substantial contributions to conception and design, Drafting the article, Critical revision of the article, Final approval of the version to be published
Alya Saif Al-Mazrouei – Substantial contributions to conception and design, Drafting the article, Critical revision of the article, Final approval of the version to be published
Faisal Mohammad Al-Badri – Substantial contributions to conception and design, Drafting the article, Critical revision of the article, Final approval of the version to be published
Guarantor of submission
The corresponding author is the guarantor of submission.
Source of support
None
Conflict of interest
Authors declare no conflict of interest.
Copyright
© 2016 Bassem Mahmoud Abou Hussein et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.



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