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CASE REPORT
A Rare Case of Perforated Tubular Ileal Duplication in 72-Year-
Old Male
Gajanan Ekbote &Ashish Balwant Pokharkar &
Prashant Moon
Received: 9 June 2011 / Accepted: 11 September 2012 / Published online: 27 September 2012
#Association of Surgeons of India 2012
Abstract Enteric duplication is a rare anomaly, which is
common in pediatric population, and it is very rare in adults.
Enteric duplication can occur anywhere from mouth to anus.
Ileal duplication is the most common (30 %) followed by
duodenum, stomach, jejunum, colon and rectum. Though
most cases are presented within the first 2 years of life, some
may present at later age. Here we report a case of a 72-year-
old male presented to us in emergency as acute abdomen,
which was presumptively diagnosed as ca rectosigmoid, but
later the patient developed perforative peritonitis. On ex-
ploratory laparotomy he was diagnosed as a case of perfo-
rated ileal duplication cyst. Though these lesions are very
rare, the importance of enteric duplication cysts lies in the
fact that they can mimic many disease conditions and may
be left untreated.
Keywords Ileum .Intestinal duplication .Perforation
Introduction
Duplication cysts are a rare group of anomalies which vary
in their presentation. Often they present in a very nonspe-
cific way like vague pain in the abdomen or lump in the
abdomen. Rarely, they present as intestinal obstruction,
perforative peritonitis, malignancy, melena, intussusception
and constipation. There are different theories regarding their
origin but none are proven yet. Enteric duplication can occur
along the entire length of alimentary tract. They are located
along the mesenteric border of the intestine and are lined by
smooth muscle. Heterotopic gastric mucosa often lines
duplication cyst, and its inflammation is responsible for
melena, perforation and intestinal obstruction. Enteric du-
plication can be a diagnostic dilemma for those who are
involved in its management. Often diagnosis is made on the
operating table as in our case.
Case report
A 72-year-old Indian male came to us with history of pain in
the abdomen for 3 months, and vomiting and loose motions
for 10 days. On admission, it was found that the patient had
tenderness in the left iliac fossa and hypogastric region, and
he was febrile with raised leukocyte count. X-ray erect
abdomen was normal. On ultrasonography, it was found that
the patient had thickened wall of the rectosigmoid junction
along with few thickened ileal loops. Patient’s CT scan
abdomen was done, which showed rectosigmoid wall thick-
ening and the hypoechoic mass attached to the rectum on the
right side. Barium enema showed the apple core sign likely
to be due to malignancy, but on colonoscopy all large colon
found to be normal.
Decision was taken for elective laparotomy, but a day
before elective surgery the patient developed severe pain in
the abdomen with rigidity. X-ray erect abdomen showed air
under the right side of the diaphragm, so emergency lapa-
rotomy was done.
During exploration, we found ruptured small bowel with
inflammatory bowel loops adhered to the rectosigmoid re-
gion in the left iliac fossa. On separating adhesions, we
found interesting finding: a 10 cm duplicated ileal loop,
which was perforated. On opening its anterior wall, it was
found to be communicating both proximally and distally
(Figs. 1,2and 3) Segmented bowel resection and end-to-
end anastomosis was performed. Patient’s postoperative re-
covery was uneventful.
G. Ekbote :A. B. Pokharkar (*):P. Moon
B. J. Medical College,
Pune, Maharashtra, India
e-mail: ashishpokharkar@rediffmail.com
Indian J Surg (June 2013) 75(Suppl 1):S418–S420
DOI 10.1007/s12262-012-0749-4
Discussion
Reginald Filtz [1] first used the term ‘intestinal duplication’
in 1844. Intestinal duplication anomalies are generally com-
mon in pediatric population within the first 2 years of life
[2]. About 2/3 cases are detected within the first 2 years.
This finding is very rare in adult population.
Several theories are proposed for the pathogenesis of
intestinal duplication. According to split notochord theory,
splitting of the notochord is a primary event during embry-
ological growth responsible for duplications. Bentley and
Smith in their landmark article described the concept of the
split notochord and found a spectrum of disorders that share
a common embryogenesis, which includes enteric duplica-
tion along with vertebral anomalies [3]. Other theories are
persistent vacuolation theory, faulty separation of the endo-
derm and notochord, localized forms of twinning, etc [4].
Duplication cysts can be found anywhere from mouth to
anus but is most common in the small intestine. Ileal duplica-
tion is the most common (30 %) followed by duodenum,
stomach, jejunum, colon and rectum [5]. There are two forms
of duplications: spherical or cystic duplications and tubular
duplications. Cystic duplications (75 %) are more common
than tubular duplications (25 %) [6]. These hollow structures
possess a well-developed smooth muscle wall and are lined by
intestinal mucosa. Often heterotopic tissues like gastric mu-
cosa or pancreatic tissue are present in the wall of them. The
length of tubular duplications varies from few centimeters up
to as long as the whole length of the small intestine. Tubular
duplication often communicates with the main intestine prox-
imally or distally or at both ends like in our case. These
duplications share common wall and blood supply.
Intestinal duplications are very difficult to diagnose preop-
eratively. They present with myriad of symptoms. The symp-
toms are often related to location of duplication. Most cases
are detected during early years of life; as far as we know there
are very few cases in the literature which are diagnosed at old
age in adults. The most common clinical presentation in adults
is vague abdominal pain and dyspepsia. Other less common
presentations are intestinal obstruction, perforative peritonitis,
malignancy [7], melena [8], intussusception and constipation.
Acid secreting heterotopic gastric mucosa often causes peptic
ulcer disease, which leads to perforation and peritonitis.
Diagnosis of intestinal duplication is often a challenging
task. Most of the time it is not possible to diagnose preop-
eratively or misdiagnosis is made like in our case and
diagnosis is made on the operating table. Various radiolog-
ical modalities like ultrasound and CT scan are helpful in
making diagnosis. Ultrasound is important in case of cystic
duplication but is often inconclusive in tubular duplications
[9,10]. Barium enemas can be helpful in few cases but most
of the time is not much helpful. 99Tcm-pertechnetate scan-
ning is a reliable 99Tcm-pertechnetate, noninvasive method
to diagnose ectopic gastric mucosa [11,12].
Most authors believe that treatment is required in symp-
tomatic patients and those who present with complications.
Intestinal resection and anastomosis is required in all cases
and include adjacent normal intestine because of common
sharing of bowel wall and of blood supply [13,14]. Postop-
erative outcomes are often favorable. Though there are some
Fig. 1 Ileal duplication (arrow) with perforation on exploratory
laparotomy
Fig. 2 Wall of intestinal duplication is cut opened to show common
sharing of wall (arrow) between duplication and normal intestine;
Ryle’s tube is within normal ileum
Fig. 3 Diagrammatic representation of ileal duplication
Indian J Surg (June 2013) 75(Suppl 1):S418–S420 S419
case reports of malignancies in ileal duplication, these find-
ings are rare [2].
To conclude, ileal duplications are rare anomalies in
adults, which are often undiagnosed preoperatively and
can present as perforative peritonitis, intestinal obstruction,
bleeding and malignancy.
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