This Article Abstract Figures Only Full Text (PDF) Services Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Lalloo, S Articles by Maharajh, J Search for Related Content PubMed PubMed Citation Articles by Lalloo, S Articles by Maharajh, J

Abdominal cocoon associated with tuberculous pelvic inflammatory disease

Department of Radiology, Nelson R Mandela School of Medicine, University of Natal, Private Bag 7, Congella 4013, Durban, South Africa

	Abstract

Top Abstract Introduction Case report Discussion References

 
Abdominal cocoon is a rare acquired condition in which there is encapsulation of the small bowel by a fibrous membrane. The authors describe a case wherein an organism was identified for the first time. The clinical, pathological and radiological features of this unusual disease are reviewed. Peritoneal encapsulation, a related subject, is also discussed.

	Introduction

Top Abstract Introduction Case report Discussion References

 
Although well documented, abdominal cocoon (AC) remains a clinical curiosity, the exact aetiology of which is unclear. We review the subject and present a case in which we believe a causative pathogen has been positively identified for the first time.

	Case report

Top Abstract Introduction Case report Discussion References

 
A 36-year-old African female presented with a 3 month history of progressive lower abdominal distension, intermittent suprapubic pain and, more recently, an offensive vaginal discharge. The last of her four children, all delivered by caesarean section, was born 3 years previously. She had amenorrhoea since using injectable contraception over the 2 years preceding presentation, following the birth of her last child. She had no history of any significant illness, surgery or prolonged medication.

At presentation she was apyrexial and normotensive. Physical examination and transabdominal ultrasound suggested a neoplasm of the left ovary. The patient was prepared for a staging laparotomy with a view to hysterectomy and bilateral salpingo-oophorectomy. Blood analysis showed a haemoglobin level of 10.7 g dl^-1 and a white cell count of 4.4 x 10⁹ l^-1. The presence of a subtle unilateral midzone pulmonary infiltrate and a small left pleural effusion on the pre-operative chest radiograph raised a suspicion of pulmonary tuberculosis. There were no cardiorespiratory symptoms, and chest auscultation was normal.

At laparotomy, pelvic adhesions were broken down, followed by the removal of an abnormally firm and bulky uterus and bilateral cystic adnexal masses, the left mass being approximately 5 cm in size and partly adherent to a bizarre fluctuant central retroperitoneal mass. The surgeon noted "absence of the small bowel with a soft, fluctuant, non-pulsatile mass in the central retroperitoneum extending to the right and having a slightly thickened opaque covering, with surface exudates". Tubercles were noted over the liver and stomach. Both kidneys were palpated. It was concluded that CT was necessary before any further surgery could be performed.

Histology confirmed caseating granulomatous inflammation of the cervix, endometrium and both fallopian tubes, with numerous acid-fast bacilli. Multiple biopsies of peritoneal tubercles and omental lymph nodes demonstrated similar findings. Both ovaries demonstrated only corpus luteal cysts.

Helical CT performed 3 days following laparotomy showed the entire small bowel contained within a sac, the wall of which was thickened and demonstrated mild enhancement. Parietal peritoneal enhancement and some post-operative ascites were also noted (Figure 1). Subsequent barium follow-through examination demonstrated constant elliptical containment of the small bowel without malrotation or obstruction (Figure 2).

View larger version (64K): [in this window] [in a new window]   Figure 1. CT demonstrating small bowel contained within a sac. (a) The compressed third part of the duodenum crossing the midline (asterisk) and (b) the thickened enhancing encapsulating membrane (short arrows) are visible.

View larger version (105K): [in this window] [in a new window]   Figure 2. 15 min barium follow-through radiograph showing eccentric elliptical clustering (arrows) of otherwise normal small bowel loops.

	Discussion

Top Abstract Introduction Case report Discussion References

 
AC is an infrequently reported clinical entity. Since the earliest description by Owtschinnikow in 1907, entitled "peritonitis chronica fibrosa incapsulata", the condition has been named differently by various authors [1]. The term "abdominal cocoon" was first applied by Foo et al in 1978 [2].

Sclerosing encapsulating peritonitis (SEP) is an acquired condition that is often idiopathic. A history of previous abdominal surgery or peritonitis, chronic ambulatory peritoneal dialysis and the prolonged use of the ß-adrenergic blocker Practolol have been implicated as causative factors [3, 4]. AC is a peculiar morphological variant of SEP in which part of, or less frequently the entire, small bowel is encased in a thick, opaque, dense fibrous membrane [5]. Geographically, AC tends to be confined to the tropics and subtropical regions and occurs mainly in young females. Retrograde menstruation coupled with viral infection, once theorized as a possible aetiology, does not explain the occasional occurrence of AC in males. Foo et al [2] postulated an endemic microbial agent, viz. tuberculosis, however no organisms were isolated in any of the previously reported cases.

Clinical presentation includes acute, subacute or chronic intestinal obstruction, abdominal distension, weight loss and a fluctuant abdominal mass. The classic barium finding, described by Sieck et al [5], of a serpentine or concertina-like configuration of dilated small bowel loops in a fixed U-shaped cluster, is not always present [6]. The CT appearance of AC has rarely been reported [1, 7]. It may demonstrate adherent small bowel loops encased within a thick enhancing peritoneal membrane. Other CT features of SEP include signs of obstruction, agglutination and fixation of intestinal loops, mural thickening, ascites and localized fluid collections, peritoneal thickening and enhancement, peritoneal or intestinal mural calcifications, and reactive adenopathy [3]. Ascites is uncommon in the endemic form of AC [5]. No peritoneal fluid accumulation was evident in this case.

Prior to the advent of CT, the correct diagnosis was usually made at surgery where the AC appears as a dense grey-white capsule containing part of, or less frequently the entire, small bowel [5]. The capsule may be adherent to the surrounding structures. When the capsule is incised, the bowel loops within are easily freed although adhesions between loops and the capsule wall may occur. Decortication is usually possible, although extensive surgery and unnecessary bowel resection are associated with a high morbidity. Symptomatic relief generally ensues following conservative surgery, although recurrence has been reported [1, 5, 6].

Histopathological examination of the encapsulating membrane constantly shows thickened vascular fibrocollagenous tissue, with or without areas of lymphocyte and plasma cell infiltrates. A covering of mesothelial cells may be found in focal areas. Mesenteric lymph nodes demonstrate non-specific reactive hyperplasia and organisms are typically absent [2, 5, 6]. Ascitic fluid analysis in a few of the previously reported cases was non-contributory.

Peritoneal encapsulation (PE) is a rare congenital anomaly which parallels SEP or AC. First documented by Cleland in 1868, it is characterized by the entire small bowel, or less likely part of it lying behind an accessory but otherwise normal transparent peritoneal membrane [8, 9]. It is generally agreed that this membrane is derived from the yolk sac as it is drawn into the embryonic abdominal cavity with the midgut behind it during the 12th week of gestation. It has been associated with midgut malrotation and a vascular anomaly [10, 11]. PE is usually asymptomatic and is discovered predominantly in older males as an incidental finding at laparotomy for other indications. Intermittent abdominal pain, acute intestinal obstruction and, rarely, aortic occlusion have been reported [11–13]. At surgery the membrane is found deep to the omentum, attached laterally to the ascending and descending colon, superiorly to the transverse mesocolon and caudally to the posterior parietal peritoneum [5]. Although easily excised, the membrane is best left alone when discovered incidentally. Histological examination reveals normal peritoneum without signs of inflammation [9].

Although excision biopsies of the encapsulating sac were not obtained in our case, the evidence presented indicates tuberculosis as the likely cause of the AC secondary to peritoneal extension of chronic pelvic inflammatory disease (PID). One may theoretically argue that the findings could represent tuberculous PID and peritonitis, complicating a pre-existing idiopathic AC or PE. The co-existence of PE and AC has been described [1].

Foo et al [2] described the presence of an epitheliod granuloma in one of their specimens. Sahoo et al [6] reported tuberculostatic treatment of an 8-year-old male for 4 months preceding the diagnosis of AC, following a high erythrocyte sedimentation rate and a positive Mantoux test. However, in both cases no acid fast bacilli were found. To our knowledge, this is the first case of abdominal cocooning where a likely causative endemic organism was confirmed on pathological examination.

Received for publication May 30, 2001. Revision received October 1, 2001. Accepted for publication October 9, 2001.

	References

Top Abstract Introduction Case report Discussion References

 

1. Tsunoda T, Mochinaga N, Eto T, Furui J, Tomioka T, Takahara H. Sclerosing encapsulating peritonitis combined with peritoneal encapsulation. Arch Surg 1993;128:353–5.[Abstract/Free Full Text]
2. Foo KT, Ng KC, Rauff A, Foong WC, Sinniah R. Unusual small intestinal obstruction in girls: the abdominal cocoon. Br J Surg 1978;65:427–30.[Medline]
3. 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 disease. Abdom Imaging 1995;20:414–20.[Medline]
4. Brown P, Baddeley H, Read AE, Davies JD, McGarry J. Sclerosing peritonitis, an unusual reaction to a ß-adrenergic blocking drug (Practolol). Lancet 1974;2:1477–81.[Medline]
5. Sieck JO, Cowgill R, Larkworthy W. Peritoneal encapsulation and abdominal cocoon: case reports and a review of the literature. Gastroenterology 1983;84:1597–1601.[Medline]
6. Sahoo SP, Gangopadhyay AN, Gupta DK, Gopal SC, Sharma SP, Dash RN. Abdominal cocoon in children: a report of four cases. J Pediatr Surg 1996;31:987–8.[Medline]
7. Wig JD, Gupta SK. Computed tomography in abdominal cocoon. J Clin Gastroenterol 1998;27:259–60.[Medline]
8. Cleland J. On an abnormal arrangement of the peritoneum, with remarks on the development of the mesocolon. J Anat Physiol 1868;2:201–6.[Medline]
9. Casas JD, Mariscal A, Martinez M. Peritoneal encapsulation: CT appearance. AJR 1998;171:1017–9.[Free Full Text]
10. Walsh TN, Russell J. Peritoneal encapsulation of the small bowel. Br J Surg 1988;75:1148.[Medline]
11. Adedeji AO, McAdam WA. Small bowel obstruction due to encapsulation and abnormal artery. Postgrad Med J 1994;70:132–3.[Abstract/Free Full Text]
12. Silva MB Jr, Connolly MM, Burford-Foggs A, Flinn WR. Acute aortic occlusion as a result of extrinsic compression from peritoneal encapsulation. J Vasc Surg 1992;16:286–9.[Medline]
13. Awasthi S, Saraswat VA, Kapoor VK. Peritoneal encapsulation of the small bowel: a rare cause of intestinal obstruction. Am J Gastroenterol 1991;86:383.[Medline]

This article has been cited by other articles:

				L. Menassa-Moussa, L. Bleibel, C. Sader-Ghorra, T. Smayra, and N. J. Aoun MRI findings in intestinal cocoon. Am. J. Roentgenol., March 1, 2006; 186(3): 905 - 906. [Full Text] [PDF]

				S. Gupta, R. G. Shirahatti, and J. Anand CT Findings of an Abdominal Cocoon Am. J. Roentgenol., December 1, 2004; 183(6): 1658 - 1660. [Full Text] [PDF]

				J. Hur, K. W. Kim, M.-S. Park, and J.-S. Yu Abdominal Cocoon: Preoperative Diagnostic Clues from Radiologic Imaging with Pathologic Correlation Am. J. Roentgenol., March 1, 2004; 182(3): 639 - 641. [Full Text] [PDF]

				S. B. Vijayaraghavan, C. Palanivelu, K. Sendhilkumar, and R. Parthasarathi Abdominal Cocoon: Sonographic Features J. Ultrasound Med., July 1, 2003; 22(7): 719 - 721. [Full Text] [PDF]

This Article Abstract Figures Only Full Text (PDF) Services Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Lalloo, S Articles by Maharajh, J Search for Related Content PubMed PubMed Citation Articles by Lalloo, S Articles by Maharajh, J