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Enteric duplication cyst in an 8 year old child

ABSTRACT

Enteric duplications are rare congenital anomalies that can occur along the entire length of the gastrointestinal system. Their mostcommon location is the small intestine, mostly the terminal ileum. Almost 70% of the cases present within 1 st year of life. Thecommon symptoms include abdominal pain, vomiting, distension and constipation. Ultrasound, computed tomography scan andmagnetic resonance imaging are useful in diagnosing the lesion. Excision of the duplication cyst is the treatment of choice. This is acase report of an 8-year-old male child who presented with enteric duplication cyst, which was attached to the terminal ileum withno communication with the bowel lumen. The cyst was excised and the child made a satisfactory recovery.

Key words: Duplication cyst, enteric duplication, ileum

IntroductionEnteric duplications are rare congenital anomaliesthat can occur a long the ent i re length of thegastrointestinal system. They are defined by theirhistological appearance, which mimics that of the nativegastrointestinal tract possessing an inner mucosa-submucosa layer surrounded by an outer smooth-muscle layer. [1] The commonest location is the smallintestine, usually the terminal ileum. The estimatedincidence is 1:4500 newborns. [2,3] Duplications of ileumappear as non-communicating spherical cysts. Theyare mistaken for mesenteric cysts, but are recognisedby mucosal lining. [4] The objective of this paper wasto discuss the clinical presentation, investigations,diagnosis and treatment of an 8-year-old child thatpresented with a duplication cyst of the ileum.

Siddharth Pramod Dubhashi 1, Rajat Dinesh Sindwani 1, Sangram Jadhav 1, Harsh Kumar 21Departments of Surgery, and 2Pathology, Padmashree Dr. D.Y. Patil Medical College, Hospital and Research Centre,

Dr. D.Y. Patil Vidyapeeth, Pune, Maharashtra, India

Address for correspondence: Dr. Siddharth Pramod Dubhashi,A-2/103, Shivranjan Towers, Someshwarwadi, Pashan,Pune - 411 008, Maharashtra, India.E-mail: [email protected]

Case ReportThis is a case report of an 8-year-old male child whopresented with recurrent colicy pain in the abdomen for3 years and periodic episodes of bilious vomiting. There

was no history of fever or any other major illness. Onexamination, there was a tense cystic mass in the rightiliac fossa with tenderness on deep palpation. No other

abnormal finding on examination.

Haematological and biochemical investigations were within the normal limits. Ultrasound of the abdomenshowed a round 2.8 cm × 2.6 cm × 1.8 cm well definedanechoic cystic structure on the right periumbilical region,

with thin septa within just beneath the subcutaneoustissue adjacent to the small bowel loops, suggestive ofmesenteric cyst or enteric duplication cyst. Computedtomography (CT) scan of the abdomen showed a smooth,rounded, fluid-filled cystic lesion with thin slightlyenhancing wall [Figure 1].

Patient had exploratory laparotomy. There was a 5 cm× 3 cm cyst, 10 cm proximal to ileo-ceacal junction withadhesions between the cyst and ileo-ceacal junction andanterior abdominal wall. The base of the cyst was attachedto the terminal ileum, but was not communicating withthe bowel lumen. The cyst was excised completely. Thecontinuity of the underlying bowel was maintained andthe seromuscular defect was closed [Figures 2 and 3].

Case Report

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Dubhashi, et al .: Enteric duplication cyst

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The cyst contained clear serous fluid. Cut section of the cystshowed small bowel mucosal lining. Microscopically, thecyst was lined by low columnar to cuboidal atrophic mucosa

with mild sub-epithelial round cell infiltration and a well-developed smooth muscle coat [Figure 4] and a histologicaldiagnosis of intestinal duplication cyst was made.

DiscussionReginald Fitz first used the word “duplication” in 1884, todescribe the remnants of the omphalo-mesenteric duct. [5] The term “alimentary tract duplication” was coined byW.E. Ladd to describe those congenital malformationsthat are seen on the mesenteric side of the associatedalimentary tract and share a common blood supply withthe native bowel. [6] The duplication is explained as a defectin the recanalization of the intestinal lumen after the solidstage of the embryological development. [4] Other theories

include-persisting embryological diverticula or “abortedGemini,” intrauterine vascular accident theory. [7] The

lesions are classified as: (1) Classical, with the sharedcommon wall and blood supply (2). Completely isolatedfrom the bowel, which has an independent blood supply(3) Intermediate, with part of the duplication independentand part of its sharing a common wall with bowel. [2] Thelesion seen in our case was of an intermediate type. Thevascular classification to facilitate surgical managementdivides the enteric duplications into - Type I (parallel) withthe duplication located on the border of one mesenteric leafand the straight artery to the duplication is separated fromthe straight artery to the bowel, Type II (intramesenteric)

with the duplication located between the two leaves of themesentery and the straight arteries pass over both surfacesof the duplication to reach the adjacent bowel. [8]

Almost 70% of the enteric duplications that present withsymptoms do so within the 1 st year of life and 85% by2 years of age. [3] The common symptoms include abdominalpain, vomiting, distension and constipation. Small cysticduplications can act as a lead point for small bowel

Figure 1: Computed tomography scan abdomen Figure 2: Duplication cyst proximal to ileo-cecal junction

Figure 3: Cut section of cyst Figure 4: Histopathological picture showing intestinal layers

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intussusception or result in localized volvulus. Largerduplications can cause compression of adjacent intestineand cause obstructive symptoms. A malignant change canoccur within the intestinal duplication. [7] Ultrasound, CTscan and magnetic resonance imaging (MRI) are usefulin diagnosing the lesion. Ultrasound shows the double

wall or muscular rim sign with an echogenic inner margincorresponding to the mucosa surrounded by a hypoechoicrim of tissue representing the smooth muscle layer. [1] Thelesion manifests as a smooth rounded, fluid filled cysts ortubular structure with thin slightly enhancing wall on CTscan. MRI scan shows intracystic fluid with heterogeneoussignal density on T1-weighted image and homogenous highsignal intensity on T2-weighted image.

Due to the possible complications such as bowelperforation, bleeding, obstruction and malignant change,intestinal duplications require surgical intervention.

Excision of the duplication is the treatment of choice.Small cystic or short tubular duplication can be managed

with segmental resection along with adjacent bowel. Longtubular duplications will require mucosal stripping througha series of multiple incisions. [9] In our case, the cyst hadno communication with the bowel lumen and hence, bowelresection was not required.

ConclusionEnteric duplication cysts are rare congenital anomaliesthat have a varied clinical presentation. Early diagnosis

and treatment reduces the risk of complications. Resectionof the duplication cyst is the treatment of choice.

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Sonographic pitfalls in the diagnosis of enteric duplication

cysts. AJR Am J Roentgenol 2005;184:521-5.2. Prada Arias M, García Lorenzo F, Montero Sánchez M,Muguerza Vellibre R. Enteric duplication cyst resemblingumbilical cord cyst. J Perinatol 2006;26:368-70.

3. Gálvez Y, Skába R, Kalousová J, Rousková B, Hríbal Z,Snajdauf J. Alimentary tract duplications in children:High incidence of associated anomalies. Eur J Pediatr Surg2004;14:79-84.

4. Chaudhary S, Raju U, Harjai M, Gupta M. Intestinalduplication cyst. Med J Armed Forces India 2006;62:87-8.

5. Bower RJ, Sieber WK, Kiesewetter WB. Alimentary tractduplications in children. Ann Surg 1978;188:669-74.

6. Ladd WE, Gross RE. Surgical treatment of duplications of thealimentary tract. Surg Gynaecol Obstet 1940;70:295-307.

7. Blank G, Königsr ainer A, Sipos B, Ladurn er R.Adenocarcinoma arising in a cystic duplication of the smallbowel: Case report and review of literature. World J SurgOncol 2012;10:55.

8. Li L, Zhang JZ, Wang YX. Vascular classification for smallintestinal duplications: Experience with 80 cases. J PediatrSurg 1998;33:1243-5.

9. Bond SJ, Graff DB. Gastrointestinal duplications. In: ONeil JA, Grosfeld JL, Tanskalsrud EW, Coran AG, editors.Paediatric Surgery. 5 th ed. Mosby; 1998. p. 1257-63.

How to cite this article: Dubhashi SP, Sindwani RD, Jadhav S, Kumar H.Enteric duplication cyst in an 8 year old child. Arch Int Surg 2013;3:226-8.

Source of Support: Nil. Con ict of Interest: None declared.

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