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Turkish Journal of Trauma & Emergency Ulus Travma Acil Cerrahi Derg 2011;17 (6):557-560 Case Report Olgu Sunumu doi: 10.5505/tjtes.2011.39018

Abdominal cocoon syndrome as a rare cause of mechanical bowel obstruction: report of two cases

Bağırsak tıkanıklığının nadir bir sebebi olan abdominal koza sendromu: İki olgu sunumu

Levent YENİAY, Can Avni KARACA, Cemil ÇALIŞKAN, Özgür FIRAT, Sinan Muhtar ERSİN, Erhan AKGÜN

An abdominal cocoon is an extremely rare condition, and Abdominal koza literatürde genelde genç adölesan kadın- has been reported mainly in young adolescent women as a larda ince bağırsak tıkanıklığının bir nedeni olarak bildirilen cause of small bowel obstruction. In these patients, the small nadir bir durumdur. Bu hastalarda ince bağırsak “abdomi- bowel is encased in a fibrous sac called an abdominal co- nal koza” olarak adlandırılan fibröz bir kese içine sarılmış- coon. We hereby present two cases who were diagnosed only tır. Biz burada tanısı ancak laparotomi ile konulabilen iki ol- by and their correlation with the literature. They guyu literatürdeki bilgilerle birlikte sunuyoruz. Her iki olgu- both received early intervention, thus preventing the need for ya da erken dönemde müdahale edilerek bağırsak rezeksiyo- . The pathology of both membranes showed nundan kaçınılmıştır. Her iki olguda da membranın patolojik inflammation. incelemesi enflamatuvar bir sürece işaret etmiştir. Key Words: Abdominal cocoon; peritoneal encapsulation. Anahtar Sözcükler: Abdominal koza; periton kapsülleme.

Abdominal cocoon syndrome describes the condi- CASE REPORTS tion of partial or total encapsulation of abdominal vis- Case 1- A 26-year-old female patient was admit- cera within a dense fibrous membrane, and was first ted to the emergency department with the complaints [1] described by Foo et al. in 1978. The majority of the of colic abdominal and persisting for cases are reported from tropical and subtropical cli- two days. There were similar episodes with spontane- mate belts of the world. To our best knowledge, the ous symptomatic relief in the detailed history of the only case reported from a non-tropical zone is from patient. Anamnesis revealed no history of chronic ill- [2] England, in which the case was born in Pakistan. ness, chronic medication or previous operation. Several theories have been suggested regarding the In the physical examination, there were signs of etiology of the disease, but the majority of the cases [3] peritoneal irritation especially in the lower quadrants are of unknown etiology. of the abdomen with rebound tenderness. No disten- Herein, we present two cases of idiopathic abdomi- tion was observed and the bowel sounds were atten- nal cocoon syndrome who were treated successfully in uated. Air fluid levels were seen in plain abdominal our surgery department, together with a brief review X-rays, and a computerized tomography revealed dis- of the literature. tended small bowels to the level of the terminal .

Department of , Ege University Faculty of Medicine, Ege Üniversitesi Tıp Fakültesi Genel Cerrahi Anabilim Dalı, Izmir, Turkey. İzmir.

Correspondence (İletişim): Can Avni Karaca, M.D. 252. Sok., No: 40 Da: 1, Izmir, Turkey. Tel: +90 - 232 - 390 50 50 e-mail (e-posta): [email protected]

557 Ulus Travma Acil Cerrahi Derg

(a) (b) Fig. 1. Case 1. (a) Bowels and the abdominal viscera were found to be under a thick and dense membrane. (b) The was firmly adhered to the cecal wall. (Color figure can be viewed in the online issue, which is available at www.tjtes.org)

Blood chemistry, total blood count and urine analysis The diagnosis was made perioperatively, and the showed no abnormality except a slight elevation of C- patient was further questioned regarding secondary reactive protein. causes of abdominal cocoon syndrome. There was no positive history of these causes, which are reviewed in The patient was then explored based on the diag- the Discussion section. nosis of . In the operational observa- tion, bowels and the abdominal viscera were found The patient was discharged on the 6th postopera- to be under a thick and dense membrane, and the ob- tive day. struction site causing the symptoms was a short small The histopathologic analysis of the specimen was bowel segment adjacent to the ileocecal valve (Fig. reported as fibrosis, inflammation and fibrin exuda- 1a). The appendix was firmly adhered to the cecal wall tion. (Fig. 1b). There was no major omentum present. The patient is doing well on follow-up, with no The patient underwent simply bridectomy; no re- symptoms of bowel obstruction. section was performed. After the incision of the mem- Case 2- A 71-year-old male patient was admitted brane, the bowels were freed, leaving no narrowed seg- to the emergency department with the complaint of ment. The postoperative course was uncomplicated. profuse vomiting after meals and absence of gas dis- The patient was allowed to feed via oral route by the charge for nearly two days. He described three similar 3rd postoperative day, and stool discharge occurred on episodes with spontaneous symptomatic relief within the 4th postoperative day. three months. He had no chronic disease.

(a) (b) Fig. 2. Case 2. A cardinal symptom of mechanical bowel obstruction. (Color figure can be viewed in the online issue, which is available at www.tjtes.org)

558 Kasım - November 2011 Abdominal cocoon syndrome as a rare cause of mechanical bowel obstruction

Palpation of the upper quadrants of the abdomen sexuality factors.[6,7] None of these secondary patho- was painful with rebound tenderness. There was a soft genetic factors was encountered in our cases. The ab- palpable mass located in the left quadrants of the ab- sence of the major omentum in both of our cases may domen. No distention was observed, and the bowel suggest the association of some unknown genetic fac- sounds were not altered. Plain X-rays of the abdo- tors that play a role in the pathogenesis of idiopathic men showed no significant pathologic sign. Abdomi- disease.[8] nal computerized tomography revealed distention of The clinical presentation of the abdominal co- stomach, and a short segment of the proxi- coon syndrome mostly occurs as acute abdomen, mal with a small amount of fluid collection which, in most cases, requires surgical intervention. around the stomach. The tomography reported a clus- Preoperative diagnosis of the syndrome is usually dif- tering of bowels to the left quadrants of the abdomen. ficult. There are four main clinical features suggested The blood chemistry and total blood count showed no in the literature by Yip and Lee[9] for preoperative di- abnormality. agnosis. These are: The patient underwent an emergency laparotomy A relatively young female patient without an obvi- with a diagnosis of acute abdomen. During the opera- ous cause of bowel obstruction. tional evaluation, the bowels were found to be encap- sulated in a thick membrane causing obstruction, but Past history of similar episodes with symptomatic preventing the distention, which is a cardinal symp- relief. tom of mechanical bowel obstruction (Fig. 2a, b). As Presentation with symptoms suggestive of bowel observed in the first case, the major omentum was obstruction but absence of cardinal symptoms such as missing. distention. The patient underwent simple bridectomy, and the Presence of soft non-tender . adhesions were freed by careful dissections in order Our first case fits most of the features above except to prevent iatrogenic bowel injuries, which are fre- for the presence of a palpable mass. The latter also fits quently reported in the literature in similar cases. The these features except for the patient being an elderly postoperative course was uneventful. Enteral feeding male patient. All these clinical features allowed us to started on the 2nd postoperative day, and stool dis- suspect the diagnosis of abdominal cocoon syndrome. charge occurred on the 6th postoperative day. This led us to decide laparotomy earlier, thus prevent- As in the first case, the diagnosis was done peri- ing strangulation and resection of the bowel. operatively. Since there was no positive history of However, the definitive diagnosis is achieved un- secondary causes, the case was admitted as primary der laparotomy. In the literature, typically, the appear- abdominal cocoon syndrome. The patient was dis- ance of the abdomen is described as the bowels be- charged on the 8th postoperative day. ing in a coil-like pattern with a dense thick membrane The histopathologic analysis of the specimen re- covering the small bowels totally or partially.[10] In vealed fibrosis and granulomatous inflammation. some distinct cases, this membrane can cover the and even solid organs. In both of our cases, Follow-up of the patient was unproblematic, with the membrane fully covered both the small and large no symptoms of bowel obstruction. intestine, but left the solid viscera uncovered.[10] DISCUSSION The treatment of choice for abdominal cocoon An abdominal cocoon was first described and syndrome is lysis of adhesions or total removal of the named by Foo et al.[1] in 1978. An abdominal cocoon is membrane. a form of intestinal encapsulation, which is mostly ob- In summary, abdominal cocoon syndrome is rare served in young girls living in tropical and subtropical and difficult to diagnose. A better awareness of the en- regions.[1,4,5] In contrast to peritoneal encapsulation, tity, plus a combination of clinical examination and the encasing membrane is an opaque fibrous structure, radiologic studies, may facilitate the preoperative di- which is not covered by a mesothelium. In the vast agnosis. Suitable and early surgery and appropriate majority of the cases, the histological evaluation of the perioperative treatment can improve patient prognosis membrane shows inflammation. in this rare condition. The pathogenesis of abdominal cocoon syndrome REFERENCES remains unknown; however, the etiopathogenesis cor- 1. Foo KT, Ng KC, Rauff A, Foong WC, Sinniah R. Unusual relates to congenital dysplasia, chronic asymptomatic small intestinal obstruction in adolescent girls: the abdomi- , some medicines (e.g., practolol), continu- nal cocoon. Br J Surg 1978;65:427-30. ous ambulatory peritoneal dialysis, and district and 2. Macklin J, Hall C, Feldman MA. Unusual cause of small

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bowel obstruction in adolescent girls: the abdominal cocoon. literature. Adv Ren Replace Ther 1998;5:157-67. J R Coll Surg Edinb 1991;36:50-2. 7. Mann RD. An instructive example of a long-latency adverse 3. Kumar M, Deb M, Parshad R. Abdominal cocoon: report of drug reaction--sclerosing peritonitis due to practolol. Phar- a case. Surg Today 2000;30:950-3. macoepidemiol Drug Saf 2007;16:1211-6. 4. Bhatnagar A, Pathania OP, Malik V, Chowdhry A. Abdomi- 8. Wei B, Wei HB, Guo WP, Zheng ZH, Huang Y, Hu BG, et al. nal cocoon causing small bowel obstruction. Indian J Gastro- Diagnosis and treatment of abdominal cocoon: a report of 24 enterol 1987;6:55-6. cases. Am J Surg 2009;198:348-53. 5. Ahmed MN, Kaur S, Zargar HU. Abdominal cocoon: an un- usual intestinal obstruction (a case report). J Postgrad Med 9. Yip FW, Lee SH. The abdominal cocoon. Aust N Z J Surg 1984;30:62-3. 1992;62:638-42. 6. Afthentopoulos IE, Passadakis P, Oreopoulos DG, Bargman 10. Sieck JO, Cowgill R, Larkworthy W. Peritoneal encapsula- J. Sclerosing peritonitis in continuous ambulatory peritoneal tion and abdominal cocoon. Case reports and a review of the dialysis patients: one center’s experience and review of the literature. 1983;84:1597-1601.

560 Kasım - November 2011