C Fotiadis, I
Papandreou, EP Misiakos, 3rd
Department of Surgery, University of Athens, School of Medicine,
M Genetzakis, GC Zografos, 1st
Department of Propedeutic Surgery, University of Athens, School of
Medicine, Hippokration Hospital, Athens, Greece
E Agapitos, Department of Pathology, University of Athens,
School of Medicine, Athens, Greece
Correspondence to: C Fotiadis, MD, Associate Professor of
Surgery, University of Athens, 8 Tripoleos Street, Melissia, Athens
15127, Greece. firstname.lastname@example.org
Gastrointestinal duplication is an uncommon congenital abnormality
in two-thirds of cases manifesting before the age of 2 years. Ileal
duplication is common while colonic duplication, either cystic or
tubular, is a rather unusual clinical entity that remains
asymptomatic and undiagnosed in most cases. Mostly occurring in
pediatric patients, colonic duplication is encountered in adults
only in a few cases. This study reports two cases of colonic
duplication in adults. Both cases presented with rectal bleeding on
admission. The study was focused on clinical, imaging, histological,
and therapeutical aspects of the presenting cases. Gastrografin
enema established the diagnosis in both cases. The cystic structure
and the adjacent part of the colon were excised en-block. The study
implies that colonic duplication, though uncommon, should be
included in the differential diagnosis of rectal bleeding.
© 2005 The WJG Press and Elsevier Inc. All rights reserved.
Key words: Colonic duplication; Gastrointestinal bleeding
Fotiadis C, Genetzakis M, Papandreou
I, Misiakos EP,
Agapitos E, Zografos GC.
Colonic duplication in adults: Report of two cases presenting with
rectal bleeding. World J Gastroenterol
2005; 11(32): 5072-5074
Gastrointestinal duplication is an uncommon congenital abnormality.
More than 80% of cases present before the age of 2 years as an acute
abdomen or bowel obstruction and can occur anywhere throughout the
Ileum is the most common site for duplication, accounting for over
60% of cases.
On the other hand, duplication of the colon is a rare abnormality,
accounting for 4-18% of all gastrointestinal duplications, often
located in the cecum[3,4].
A thorough review of the international literature since 1950, has
revealed 83 cases of colonic duplications reported up-to-date.
Although intestinal duplications are considered
to be benign lesions, mostly asymptomatic, they may result in
significant morbidity and mortality, if left untreated.
Indications for surgical intervention often arise in an acute
setting, in the form of a complication. Specifically, patients with
previously undetected duplications may present in the setting of
bowel obstruction or severe gastrointestinal hemorrhage (i.e.,
ulcerating gastric mucosa within a duplication cyst).
If encountered incidentally, these lesions should be surgically
addressed to avoid any future complication, including the
possibility of malignant degeneration within the duplication cyst[3,5].
The cases mentioned in the present article refer
to patients manifesting bleeding of the lower gastrointestinal
tract, which was finally proved as originating from the mucosa of a
A 53-year-old male was admitted to our department with rectal
bleeding. He claimed of stool mixed with blood, without pain or
changes in bowel habits, 5 d prior to admission. The patient had a
medical history of constipation, being treated with diet for more
than 20 years. On admission, he was in a stable clinical condition,
with normal vital signs and blood tests were within normal ranges.
Rectal examination confirmed the presence of blood in the stools.
Plain X-rays of the abdomen showed no particular findings.
Colonoscopy was performed showing only two small
diverticula in the sigmoid colon, but could not reveal the site of
bleeding. It was not possible for the right colon to be examined
thoroughly, due to imperfect bowel cleansing. As a second step of
the diagnostic procedure, radiographic colon imaging after
gastrografin enema was performed. It showed a fistula in the middle
of the ascending colon, communicating with a cystic formation
approximately 2 cm×1 cm in dimension. Furthermore, a computerized
tomography of the abdomen was performed, which confirmed the
findings and a possible diagnosis of colonic duplication of
ascending colon was concluded. A second colonoscopy detected the
mucosa of the colonic duplication to be the origin of bleeding.
Excision of the cystic lesion was decided (Figure
1). It was resected en-block to the section of the ascending colon
and an end-to-end anastomosis of the colon was performed.
Pathological examination of the resected lesion revealed a
duplication cyst in the mesenteric border of the ascending colon,
with normal intestinal mucosa lining its wall (Figure 2). Within the
mucosa of the duplication, a site of angiodysplasia was detected as
the origin of bleeding.
The patient had an uneventful postoperative
course and was discharged on the 6th
post-operative day. He remained asymptomatic ever since.
Figure 1 Surgical
specimen including the ascending colon together with a duplication
cyst attached to the mesenteric border of the colon.
Figure 2 Normal
intestinal mucosa of the cyst in the first case (HE ×125).
A 45-year-old male was referred to our department claiming of
fatigue and a 5-d history of bloody stools. Physical examination as
well as blood investigations were entirely unremarkable. The patient
was submitted to rectal examination as well as colonoscopy, which
did not reveal any source of bleeding. The radiological imaging
control using gastrografin enema showed a cystic formation of 0.5 cm×5
cm in size, located in the mesenteric border of the descending
colon. This finding was confirmed by a second colonoscopy, which
established the diagnosis of a colonic duplication.
A surgical intervention was decided in order to
detect the site of bleeding, as well as to prevent any further
complication of the cyst. The cystic lesion was resected en-block to
the adjacent part of the colon (Figure 3) and an end-to-end
anastomosis was performed. The cyst was confirmed to be a colonic
duplication of normal colonic mucosa according to the pathologic
study. Indeed, there was a mucosal ulceration in the cystic cavity,
probably representing the origin of bleeding. There was no immediate
or delayed post-operative complication.
Figure 3 Surgical
specimen in the second case including the descending colon with a
duplication cyst adjacent to the colon.
Intestinal duplication is a congenital malformation, that occurs
mostly in pediatric patients[2,6].
It could be encountered anywhere throughout the gastrointestinal
tract, from the mouth to the anus. Current nomenclature relies on
the anatomic location of the duplication in relation to the normal
intestinal tract and not on the histological features of the mucosal
Intestinal duplications are located
in, or attached (share a common wall) to the wall of an
adjacent part of the gastrointestinal tract and possess at least one
exterior layer of smooth muscle and are lined with various types of
Further characterization defines these abnormalities as either
spherical or tubular. Rarely, complete duplication of the colon may
Approximately 75% of duplications have been
reported to be located within the abdominal cavity. Jejunal and
ileal lesions are the most commonly encountered (53%), while colonic
lesions are found in 13% of cases. These lesions may be cystic (75%)
or tubular (25%) in appearance and characteristically arise from the
mesenteric border of the bowel[5,6].
They may or may not have one or more direct communications with the
adjacent part of the bowel across the common septum.
Non-communicating duplications typically contain clear alkaline
fluid, except in cases in which gastric mucosa is present (25%) and
acidic fluid is observed. In addition, non-activated pancreatic
enzymes may also be observed in cases of ectopic pancreatic tissue
within the duplication lesion.
In adults, either cystic or tubular duplication of the colon is a
Most colonic duplication cysts are asymptomatic
and remain undiagnosed for years.
If symptomatic, they manifest with obstruction[10,11],
Cystic duplications have been reported to cause obstruction of the
colon as a result of direct compression, volvulus, or
intussusception, whereas tubular duplications of the rectum have
been described as having direct communication with the perineum[3,13].
Although many duplications are diagnosed incidentally, most patients
present with a combination of pain and/or obstructive symptoms[14,15].
These symptoms may be the direct effects of distension of the
duplication or caused by compression of adjacent organs (including
their associated blood supplies). In addition, abrupt hemorrhage
with hemodynamic instability can be encountered in the case of a
cyst, lined with mucosa that ulcerates and eventually erodes into
adjacent organs and/or vessels[3,13].
There are scattered reports of intestinal carcinomas found within
No significant difference in clinical presentation has been detected
between communicating and non-communicating cysts.
Histological analysis typically reveals at least
one outer muscular layer with an inner gastrointestinal mucosal
lining. The mucosal lining does not necessarily correspond to that
of the adjacent normal intestine and may be comprised several
different types of gastrointestinal mucosa.
Because of the relative scarcity of such abnormalities, current
literature consists mainly of small populations and case reports
rather than any large single or multi-institutional series[8-10,13].
The first patient reported in this study,
presented with bleeding in the stools and constipation as his major
complaints on admission, while the other one claimed of rectal
bleeding. Abdominal X-rays in both cases showed no abnormal
findings. The use of contrast material in the imaging control was
necessary to discover the cysts. Ultrasonography could help in the
Colonoscopy and CT imaging could establish the diagnosis.
Colonoscopic examination revealed a cystic formation, lined by
normal intestinal mucosa, communicating with the colon. CT imaging
showed a cystic mass attached to the colon.
Preoperative diagnosis of colonic duplication is
often difficult. Symptoms usually include abdominal pain, often
confusing colonic duplication with other more common diagnoses.
Plain X-rays of the chest and abdomen should be routinely performed;
however, because of the non-specificity of their results, it is
difficult to make a pre-operative diagnosis on the basis of
radiographic findings. Computerized tomography of the chest or
abdomen is useful in establishing a diagnosis of alimentary tract
duplication during the pre-operative workup and may be used to
evaluate the synchronous lesions, once a single duplication is
Ultrasonography is also helpful in establishing a pre-operative
Treatment is reserved for symptomatic cases and
usually includes resection of the cyst and the neighboring part of
Complications related to surgical intervention are typically
nonspecific and include post-operative bleeding, infection, and
However, in patients with large tubular duplications, injury to the
normal intestine with resultant short bowel syndrome must be
In most instances, cystic duplications can be
completely excised. Resection of normal intestine must often
accompany removal of the lesion, because of the intimate attachment
of the common wall or because isolated resection of the cyst would
compromise blood flow to the adjacent intestinal segment[3,11,13,19].
An alternative approach involving marsupialization of the cystic
structure consists of a partial cystectomy combined with mucosal
stripping of the remaining cyst wall to preserve normal anatomy.
Although current literature does not specifically address the
prognosis and outcome related to the diagnosis of alimentary tract
duplications, the overall outcome is generally favorable[2,3].
RI. Gastrointestinal duplications: clinical, pathologic,
etiologic, and radiologic considerations.
Radiographics 1993; 13:
J, Rowe M. Duplications of the gastrointestinal tract. In:
Essentials of Pediatric Surgery. St. Louis, Mo.
Mosby Yearbook 1995: 520-525
GW, Gheissari A, O’Neill JA Jr. Surgical management of
alimentary tract duplications. Ann Surg
1989; 209: 167-172
PS, Nguyen LT, St-Vil D, Flageole H, Bensoussan AL, Nguyen VH,
Laberge JM. Gastrointestinal
duplications. J Pediatr
Surg 2003; 38: 740-744
CC, Yeh DC, Wu CC, Li MC, Kwan PC. Huge cystic duplication of
the ascending colon in adult. Zhonghua Yixue
Zazhi 2001; 64: 174-178
DC, Demetriades DM. Total tubular duplication of the colon and
distal ileum combined with
surgical management and long-term results. Eur J Pediatr Surg
1996; 6: 243-244
7 Li L, Zhang
JZ, Wang JZ, Wang YX, Chen RR, Chin H.
Complete duplication of the colon: definitive
by resection of the
duplication without the normal
bowel. Chin Med J 2002; 115: 779-781
G, Houry S, Huguier M. Sigmoid duplication in an adult. J
Chir 1986; 123: 169-170
BE, Franciosi RA, Akers DR. Enteric duplications: thirty-seven
cases-a vascular theory
of pathogenesis. Am
J Dis Child 1971; 122:
MI, Marks CG, Cook MG. An unusual presentation of intestinal
duplication with a literature review. Dig Dis
Sci 1996; 41:
Elst F, Hubens A. Duplication of the colon in the adult.
Acta Chir Belg 1978; 77: 335-342
JL. Diverticula and duplications of the intestinal tract. Arch
Pathol 1944; 38: 132-140
P, Costa G, Zanella L, Sguazzini G, Rossi FS, Fritelli P, Costa
G, Zanella L, Sguazzini G, Rossi FS.
in the adult. A case report of colonic duplication and a review of
the literature. Chir Ital 2002;
CK, Frizelle FA. Giant colonic diverticulum: report of four
cases and review of the literature. Dis Colon
Rectum 1998; 41:
II, Modelli ME, Pereira CR. Tubular duplication of the colon: a
case report and review of the literature.
J Pediatr 1996; 72:
H, Iwasaki I, Takahashi H. Carcinoid in a gastrointestinal
duplication. J Pediatr Surg 1986; 21: 902-904
H, Sample WF, Hasen G, Robinson JS, Sarti D. Ultrasonic
evaluation of abdominal gastrointestinal
tract duplication in
children. Radiology 1979; 131: 191-194
CK Jr. Ultrasonography of the gastrointestinal tract in infants
and children. Abdom Imag 1996; 21: 9-20
J, Ambrosetti P, Widgren S, Rohner A. Perforated tubular
duplication of the sigmoid colon in
Clin Biol 1990; 14: 776-779
Editor Wang XL and Guo SY Language
Editor Elsevier HK