Indian J Pediatr DOI 10.1007/s12098-013-1290-6

SCIENTIFIC LETTER

Cecal Duplication Cyst Presenting as a Recurrent Intestinal Obstruction T. Renu Kumar & B. Satya Srinivas

Received: 20 June 2013 / Accepted: 28 October 2013 # Dr. K C Chaudhuri Foundation 2013

To the Editor: A 45-d-old, term female baby, weighing 3.4 Kg, presented with acute intestinal obstruction without any bleeding per rectum for 3 d. Baby passed meconium on day 3 of life. Since then baby was repeatedly hospitalized for recurrent subacute intestinal obstruction. Distended abdomen with visible peristalsis in a stepladder pattern was obvious (Fig. 1a). Per rectal examination was normal. Abdominal X-ray (AXR) suggested an ileocecal obstruction and showed a radiopaque shadow in the right iliac fossa (Fig. 1b). Abdominal ultrasound (USG) suggested an ileocolic intussusception. Laparotomy revealed ileocecal obstruction with enlarged cecum with a tense cystic luminal mass and grossly dilated ileum and narrow ascending colon (Fig. 2a). Ileocecal resection with ileoascending anastomosis was performed. Gross specimen revealed a 7 × 5 × 4 cm, tense luminal cyst filled with mucoid material, arising from the mesenteric side of the cecum and extending into the ileum (Figs. 2b and 3). Microscopy showed denuded colonic epithelium with two distinct muscle layers with focal attenuation and presence of ectopic gastric mucosa suggestive of an enteric duplication cyst (Fig. 4a and b).

T. Renu Kumar (*) Department of Pediatric Surgery, Vaatsalya Hospital, Vizianagaram, Andhra Pradesh 535002, India e-mail: [email protected] B. Satya Srinivas Department of Pathology, Vaatsalya Hospital, Venkata Padma Healthcare Complex, Three Lamps Junction, Vizianagaram, Andhra Pradesh, India

Recurrent subacute intestinal obstruction in infants is usually caused by a decompressing Hirschsprung’s disease or recurrent intussusceptions. Duplication cysts (DC’s) rarely do so. DC’s are commonly located around the ileum and cecal DC’s are extremely rare [1]. We report a cecal DC presenting as recurrent intestinal obstruction causing diagnostic dilemma. Cecal DC’s can present with an abdominal mass, palpable intussusception [2, 3]; intestinal obstruction from intussusception or extrinsic compression [2]; gastrointestinal hemorrhage or abdominal pain mimicking appendicitis [4]. Like in this case abdominal distension may hinder palpation of a cystic mass. Cystic variants of DC’s lack luminal communication so present early with their mass effect. Like this case, majority present during first 3 mo of life. Persistence of a vacuole in the submucosal plane during the recanalization of the “solid stage,” of the gut can result in the development of cecal DC (The Aberrant luminal recanalization theory). Recurrent obstructions in our case were due to the initial slow enlargement of the cyst to just occlude the ileocecal valve and then ileum overcoming the obstruction by vigorous peristalsis to settle the obstruction but only to recur again and in the process ileum got significantly dilated. This time, complete occlusion of the cecum by enlarging cyst has culminated in frank obstruction. DC is ‘the most important’ differential for intussusception as both can present as mass per abdomen, both can have bleeding per rectum and both can mimic each other on USG and cologram. DC can lead to intussusception and both might co-exist [2, 5]. Like in this case AXR may show a DC as a round radio-opaque shadow. Preoperative diagnosis of enteric DC may be difficult. USG can misdiagnose a DC as intussusception as ‘bowel in bowel’ or ‘double wall’ sign may be seen in both. Hence like this case, DC should always be

Indian J Pediatr Fig. 1 a Clinical photograph showing grossly distended abdomen with visible peristalsis in a ‘step ladder pattern’. b Abdominal X-ray showing multiple dilated jejunal and ileal loops, and a well defined radiopaque shadow in the right iliac fossa with absence of colonic gas

Fig. 2 a Cecum grossly enlarged with a large cystic luminal mass causing complete ileocecal obstruction. Ascending colon is narrow and collapsed, and the ileum is 4 times that of ascending colon. b Tense luminal cystic mass extending into ileum

Fig. 3 Gross specimen with luminal cystic mass, arising from the mesenteric side of the cecum and extending into the ileum. Cyst was located sub-mucosally and filled with whitish mucoid material

Indian J Pediatr Fig. 4 Histopathology of the enucleated cyst showing denuded colonic epithelium (a) and ectopic gastric mucosa (b) with two distinct muscle layers which were attenuated at places suggestive of an enteric duplication cyst

considered in cases of intussusception or recurrent intestinal obstruction especially in younger infants. Acknowledgments The authors would like to thank Madhavi Chowdary, Clinical Assistant, Pediatric Surgery at Venkat Padma Hospital, Vizianagaram for literature search and correction of the manuscript. Contributions TRK: Patient management, manuscript preparation, final drafting. SS: Histopathological diagnosis, final manuscript drafting. TRK will act as guarantor for this paper. Conflict of Interest None. Role of Funding Source None.

References 1. Choong CK, Robertson RW, Beasley SW, Frizelle FA. Congenital caecal cystic duplication presenting with caecal volvulus in elderly woman. Int J Colorectal Dis. 1997;12:256–8. 2. Rizalar R, Somuncu S, Sözübir, Yildiz L, Gürses N. Cecal duplications: A rare cause for secondary intussusception. Indian J Pediatr. 1996;63:563–6. 3. Bower RJ, Sieber WK, Kiesewetter WB. Alimentary tract duplications in children. Ann Surg. 1978;188:669–74. 4. Ildstad ST, Tollerud DJ, Weiss RG, Ryan DP, McGowan MA, Martin LW. Duplications of the alimentary tract. Ann Surg. 1988;208:184–9. 5. Backer TD, Maillet B, Debing E, Dugardeyn C, Van Den Borre C. Cystic duplication of the cecum presenting with intussusception in a 7-day-old boy. Pediatr Surg Int. 1993;8:74–7.

Cecal duplication cyst presenting as a recurrent intestinal obstruction.

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