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CASE REPORT Table of Contents   
Year : 2009  |  Volume : 52  |  Issue : 3  |  Page : 397-399
Long segment ileal duplication with extensive gastric heterotopia


1 Department of Pathology, Christian Medical College & Hospital, Ludhiana, India
2 Department of Plastic Surgery, Christian Medical College & Hospital, Ludhiana, India

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Date of Web Publication12-Aug-2009
 

   Abstract 

Duplications of the alimentary tract are rare congenital anomalies which can be found at all levels of the alimentary tract. Majority of the duplications present as spherical cysts and usually range from a few millimeters to less than ten centimeters in size. Duplications produce complications such as intestinal obstruction or hemorrhage. A two-month-old infant presented with recurrent episodes of bleeding per rectum. Laparotomy revealed a giant ileal duplicated bowel segment which exhibited extensive gastric heterotopia with focal ulceration.

Keywords: Gastrointestinal duplication, gastric heterotopia, ileal duplication

How to cite this article:
Jacob S, Mani A, Singh V P, Bhatti W. Long segment ileal duplication with extensive gastric heterotopia. Indian J Pathol Microbiol 2009;52:397-9

How to cite this URL:
Jacob S, Mani A, Singh V P, Bhatti W. Long segment ileal duplication with extensive gastric heterotopia. Indian J Pathol Microbiol [serial online] 2009 [cited 2018 Aug 22];52:397-9. Available from: http://www.ijpmonline.org/text.asp?2009/52/3/397/55006



   Introduction Top


Gastrointestinal tract duplications are uncommon congenital abnormalities with an incidence of one in 4500 births. [1] They are, by definition, located in or adjacent to the wall of the gastrointestinal tract, have smooth muscle in their walls and are lined by alimentary tract mucosa. [2] Majority of the duplications are diagnosed by two years of age. [3] Duplications may initially present with symptoms of bleeding or obstruction or they may be asymptomatic and discovered incidentally. [4] The risk of peptic ulceration due to presence of ectopic gastric mucosa and the rare occurrence of malignant transformation within the duplication remain secondary therapeutic concerns. [5],[6]


   Case Report Top


A two-month-old male baby was brought to the casualty with bleeding per rectum of one day duration. Clinical examination revealed no abnormality. X-ray abdomen was normal. Meckel's scan showed large irregular tracer uptake in the right lower abdomen. The baby was suspected to have Meckel's diverticulum or intussusception and was operated upon. Intraoperatively a 38 cm long para-mesenteric, tubular communicating duplication segment was seen 20 cm proximal to the ileo-cecal junction. The segment was resected and an end to end anastomosis was done.


   Pathology Top


The duplicated portion of the small intestine measured 38 cm in length [Figure 1]. The serosa was grossly unremarkable. On cutting open, the duplicated (mesenteric) segment was dilated, with thickened and granular mucosa and an ulcer measuring 1.2 0.2 cm. This segment was seen to be communicating at both its ends with the ileum. The normal segment (antimesenteric) showed normal intestinal folds.

Microscopic examination revealed a common muscular wall shared between the duplicated segment and the definitive ileum. The muscle layer was extremely attenuated and lost at places. The antimesenteric segment was lined by small intestinal mucosa which was unremarkable. Almost the entire duplicated (mesenteric) loop showed lining by gastric type of mucosa which in many areas showed specialized body type glands as well. The surface epithelium of the gastric mucosa was predominantly villiform [Figure 2]. The ulcer showed unhealthy granulation tissue layered over with necrotic debris. No H. pylori could be identified in the areas with gastric type of mucosa. No focus of intestinal metaplasia was detected in the ectopic gastric mucosa with Alcian blue-periodic acid Schiff staining. A final diagnosis of ileal duplication with gastric heterotopia and peptic ulceration in the duplicated segment was made.


   Discussion Top


Alimentary tract duplications are rare congenital anomalies that can occur in any portion of the gastrointestinal tract from the mouth to the anus. William Ladd was the first to coin the phrase 'duplication of the alimentary tract' in 1937 for anomalies which had a well-developed coat of smooth muscle, an epithelial lining from the alimentary tract and an attachment to some part of the alimentary tract. [4]

The etiology of alimentary tract duplications has not been well-characterized. Hypotheses have included intrauterine vascular accidents, abortive attempts at twinning, persistence of embryonic diverticulum or from incomplete recanalization, and the split notocord theory. [1],[2],[4],[7]

Although they can be encountered in patients of any age, majority of the duplications are found in infants or children, with a slight predominance in males. [1],[2],[3],[7] A few patients with alimentary tract duplications have associated congenital malformations of the skeletal or the genitourinary system. [3],[7]

Small-bowel duplications frequently present with vomiting, abdominal distension/pain or the presence of an abdominal mass, which may be due to intestinal obstruction secondary to volvulus or intussusception. [1],[2] Duplications with gastric heterotopia are liable to present with recurrent abdominal pain or rectal bleeding as a result of peptic ulceration or viscus perforation. [5],[8] The present case also presented with bleeding per rectum secondary to peptic ulceration in the duplicated segment.

Most studies report the ileum to be the favored location for duplications of the alimentary tract accounting for 30 - 60% of the lesions. [1],[2],[7] However, according to some other studies, the cecum is a more common site of duplication. [3] Duplications can be cystic or tubular. Spherical cysts constitute 82% of gastrointestinal tract duplications and are prevalent at all levels of the gastrointestinal tract. This type usually does not communicate with the lumen. Tubular variants constitute 18% of the cases and are most often encountered in small and large bowel. They frequently communicate with the lumen of the adjacent gut or outside the genitourinary system. [2] The length of the duplicated segment is less than 10 cm in 90% of the cases; rarely it can go up to 70 cm. [9] The duplicated intestinal segment is located on the mesenteric border of the intestine in contrast to Meckel's diverticulum which is seen on the antimesenteric side of the bowel. [3],[5]

Ectopic tissue is present in 30% of all the duplications, and can be comprised of gastric, squamous, transitional or ciliated mucosa or pancreatic tissue. [2],[3],[4]

A small proportion of alimentary tract duplications which persist into adult life can undergo malignant transformation. These cases show predominance in females, mostly occur between 35 to 65 years of age and arise more commonly in large-bowel duplications. The resultant tumors are usually adenocarcinoma of colonic or gastric type and rarely squamous carcinoma. [6],[10]

This case of intestinal duplication is unusual because it involved a long segment and exhibited extensive gastric heterotopia with focal peptic ulceration accounting for the main symptomatology.

 
   References Top

1.Puligandla PS, Nguyen LT, St-Vil D, Flageole H, Bensoussan AL, Nguyen VH, et al . Gastrointestinal duplications. J Pediatr Surg 2003;38:740-4.  Back to cited text no. 1  [PUBMED]  [FULLTEXT]
2.Macpherson R. Gastrointestinal tract duplications: c0 linical, pathologic, etiologic and radiologic considerations. RadioGraphics 1993;13:1063-80.  Back to cited text no. 2    
3.Ildstad ST, Tollerud DJ, Weiss RG, Ryan DP, McGowan MA, Martin LW. Duplications of the alimentary tract. Clinical characteristics, preferred treatment and associated malformations. Ann Surg 1988;208:184-9.  Back to cited text no. 3    
4.Shew SB, Holcomb GW. Alimentary tract duplications. In: Aschcraft KW, Holcomb GW, Murphy JP, editors. Pediatric Surgery. 4 th ed. Philadelphia: Elsevier Saunders; 2005. p. 543-52.  Back to cited text no. 4    
5.Duffy G, Enriquez AA, Watson WC. Duplication of the ileum with heterotopic gastric mucosa, pseudomyxoma peritonei and nonrotation of the midgut. Gastroenterology 1974;67:341-6.  Back to cited text no. 5  [PUBMED]  
6.Orr LM, Edwards AJ. Neoplastic change in duplications of the alimentary tract. Br J Surg 1975;62:269-74.  Back to cited text no. 6    
7.Favara BE, Franciosi RA, Akers DR. Enteric duplications. Thirty seven cases: A vascular theory of pathogenesis. Am J Dis Child 1971;122:501-6.  Back to cited text no. 7    
8.George RK, Kaur N, Minocha VR. Ileal duplication presenting with peptic ulcer perforation and malena. Indian J Surg 2004;66:106-8.  Back to cited text no. 8    
9.Balen EM, Hernandez-Liozain JL, Pardo F, Longo JM, Cienfuegos JA, Alzina V. Giant jejunoileal duplication: p0 renatal diagnosis and complete excision without intestinal resection. J Pediatr Surg 1993;28:1586-8.  Back to cited text no. 9    
10.Babu MS, Raza M. Adenocarcinoma in an ileal duplication. J Assoc Physicians India 2008;56:119-20.  Back to cited text no. 10  [PUBMED]  

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Correspondence Address:
Sunitha Jacob
Department of Pathology, Christian Medical College and Hospital, Ludhiana - 141 008, Punjab
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.55006

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    Figures

  [Figure 1], [Figure 2]

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    Abstract
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