New Ropanasuri J Surg.

2018;3(2):e239 Putranto et al
http://dx.doi.org/10.13181/nrjs.v3i2.56

Intestinal Duplication Management in Adult Patients: A Systematic Review

Agi S. Putranto,1 Sun P. Siregar,2 Taufik B. Satrio.3

1) Division of Digestive Surgery, 2) Training Program in Surgery, Department of Surgery, Faculty of Medicine,
Universitas Indonesia, dr Cipto Mangunkusumo General Hospital Jakarta, 3) Department of Surgery, Tangerang
General Hospital, Banten.

Abstract

Introduction. Gastrointestinal duplication is a rare congenital abnormality found in adults. This

intestinal duplication may be associated with complications i.e. perforation, bleeding, obstruction
and malignancy. Degeneration to malignancy is a basic consideration to treat a radical surgery,
which is preventive. This systematic review aims to provide evidences to evaluate the preventive or
conservative surgery in the management.
Method. A systematic review conducted according to preferred reporting items for systematic
review and meta–analysis protocols (PRISMA). Literature search proceeded on PubMed and
ScienceDirect using keywords "malignancy arising from adult duplication gastrointestinal "AND"
treatment ". All articles are selected based on inclusion and exclusion criteria. The data evaluated
included postoperative complications, recurrence, follow–up and recovery.
Results. There were eleven articles – which were case reports – analyzed. These articles reported
preventive surgery provide a better outcome than conservative one.
Conclusion. Case reports in the study showed that 63% of intestinal duplication were developed to
malignancy, requiring preventive surgery (level of evidence IV). However, this should be judge with
a careful clinical assessment (New Ropanasuri J Surg.2018;3(2):e239).
Keywords: Malignancy arising from adult duplication gastrointestinal; gastrointestinal
duplication in adult; treatment

Introduction vomiting, abdominal pain, abdominal

mass, intussusception, which can be
Gastrointestinal duplication is a accompanied by congenital
congenital abnormality found in 65–85% abnormalities. 1

two–years–old children.1 Most of these

The occurrence of intestinal duplication as
cases occur in ileocaecal, with the most
the cause of intussusception in adult is an
common location in the ileum (30%).
unusual pathologic condition which is
Duplication can be in the form of a cyst
rarely found.2 In adults, the clinical
(>85%) or tubular (10–15%) and is
manifestations of intussusception are not
located on the side of the mesentery.
typical as in pediatric and may associated
Clinical manifestations of intestinal
with complications such as intestinal
duplication in children can vary, in the
perforation, GI bleeding, intestinal
form of symptoms of obstruction, nausea,
obstruction and degeneration to
malignancy.
Correspondence The diagnosis is seldom be established
Agi S. Putranto, MD.
Department of Surgery, Faculty of Medicine
preoperatively, as the patient frequently
Universitas Indonesia admitted due to strangulated intestinal
dr. Cipto Mangunkusumo General Hospital, Jakarta
Indonesia obstruction requiring immediate surgical
Email: Agi_digestive@yahoo.com intervention. The decision of appropriate

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surgical intervention should be made,
particularly when such anomaly is found
intraoperatively. There are two
approaches in the management, namely
preventive surgery or conservative one.
Preventive means radical surgery as there
a great possibility to develop malignancy,
while as conservative means the
management with a goal to overcome the
intussusception and conserved the
intestine physiologically. As there were
lack of data, then a systematic review was
conducted to find out the highest
evidence. A case report is provided to
draw the clinical problem.
Case report
Male of 24–years–old came to emergency
department of Tangerang General
Hospital with the symptoms of intestinal
obstruction and colicky pain at the right
lower abdomen quadrant and history of
currant jelly stool for two days. No history
of previous intestinal obstruction.
On physical examination, patient was
found alert, tachycardia with normal
breath per minutes and normothermia. No
anemic nor icteric. Distended abdomen
found with tenderness at the lower right
quadrant. Signs of peritonitis were absent.
Leukocytosis found on peripheral blood
specimen, and abdominal x–rays
indicating intestinal obstruction at upper
GI level. On the laparotomy, dilated ileum
was noted, and intussusception in the
jejunum 300 cm orally to ligament of
Treitz. Resection was decided with end to
end ileo–ileal anastomosis. When
intussusception was cut, a tubular mass
measuring 14 cm long and 2 cm in
diameter with hematoma, necrotic tissue
and mucus filled the lumen was
macroscopically shown.
Microscopically, mucosa of the ileum and
the area of duplication have a complete
muscular layer, with bleeding and necrotic
Putranto et al
area involves all intestinal layers. There
was no sign of malignancy. The patient
discharged on the sixth postoperative day
and came for follow up seven days after
being discharged; no complain and he
proceeded normal activities.
Methods
A systematic review conducted to find out
the management of intestinal duplication
that have a great potency to malignant
degeneration, as found in adult. The study
proceeded in accordance with preferred
reporting items for systematic review and
meta–analysis protocols (PRISMA). The
literature search proceeded on PubMed
and ScienceDirect using keywords
"malignancy arising from adult
duplication gastrointestinal" AND
"treatment". All articles discussing the
management of intestinal duplication in
adult cases in the last ten years using
English or Indonesian were included in
this study. Those discussing degeneration
to malignancy were included. While as
those in pediatric, correspondence, or did
not mention the outcomes of the treatment
were excluded from the study. These
articles were critically appraised using
critical appraisal tool for systematic
review. Relevant data to research question
were analyzed in those articles of the
highest level of evidence.
Results
There were total of 133 articles available,
comprised of 88 articles in PubMed and
24 articles in ScienceDirect. On filtering,
eleven articles were in lined to the criteria.
In a critical review of patient’s
information, there were no information
about demographic characteristics in all
articles, but the symptoms of obstruction
including medical history and history of
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illness related to the symptoms. In the
diagnosis, the method used to diagnose
were described, including the use of
imaging (ultrasound, CT scan and MRI).
Each of article describing the
interventions that were carried out,
changes in interventions due to different
intraoperative findings or difficulties
enfaced. Reoperation for recurrence
completed with chemotherapy were
described. These eleven articles also
discussed the evaluation including clinical
assessment before and after the
intervention. The compliance of follow–
up were the focus on each article. The
outcome of overall treatments was
addressed in these articles.
Figure 1. Diagram of literature search on intestinal tuberculosis
Putranto et al
In the discussion, no article explaining the
strength and weakness of the intervention
carried out, but comparations to other
studies and the rationale as well as the
benefits of interventions were addressed.
Not a single article describing the
prognosis and patient’s perspectives. Five
articles reported preventive surgeries, and
four articles (80%) reporting recovery as
the outcome, an article (20%) reporting
recurrent and mortality. Other six articles
proceeded conservative surgery and found
four articles (50%) associated with
mortality and two articles (33%) recovery,
and three articles (67%) associated with
recurrent (table 1 and 2).
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Table 1. Data extraction preoperatively patients with intestinal duplication

Age (years)/ Surgical Type of Level of

Article Study design Initial symptom Imaging Histology
gender management duplication evidence

Barussaud (2008) Case report 67 years/M Abdominal mass Abdominal CT scan Preventive Cyst Adenocarcinoma 4
Fletcher (2012) Case report 28 years/M Abdominal pain USG, abdominal CT Conservative Cyst Adenocarcinoma 4
scan
Dalia (2017) Case report 61 years/M Abdominal mass Abdominal CT scan Conservative Cyst Transitional carcinoma 4
GIST
Cabrera (2016) Case report 71 years/F Abdominal mass Abdominal CT scan, Conservative Cyst 4
endoscopy Adenocarcinoma
Zhu (2015) Case report 62 years/M Abdominal pain Abdominal CT scan Preventive Cyst Adenocarcinoma 4
43 years/M Abdominal pain Abdominal CT scan Preventive Cyst Adenocarcinoma
73 years/M Distended abdomen Abdominal CT scan Preventive Cyst Adenocarcinoma
Yamasaki (2016) Case report 42 years/F Abdominal mass Abdominal CT scan Conservative Cyst Adenocarcinoma 4
Habib (2009) Case report 38 years/M Abdominal pain Abdominal CT scan Preventive Cyst Adenocarcinoma 4
Damiano (2011) Case report 88 years/F Hematochezia Abdominal CT scan, Conservative Tubular 4
colonoscopy Adenocarcinoma
Hee (2012) Case report 41 years/M Abdominal mass Abdominal CT scan Conservative Cyst Non–malignant 4
Kenoki (2015) Case report 20 years/M Abdominal pain Abdominal CT scan Preventive Cyst, 4
tubular Non–malignant
Kamal (2016) Case report 20 years/M Abdominal pain USG, abdominal CT Preventive Cyst 4
scan

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Table 2. Outcome of management of intestinal duplication

Article Age(years) / Postoperative Postoperative Level of

Study design Follow up Result
gender complication intervention evidence
Barussaud (2008) Case report 67 years/M Liver metastasis Abdominal CT scan Chemotherapy Died 4
Fletcher (2012) Case report 28 years/M Recurrent obstruction Abdominal CT scan Relaparotomy mass excision Died 4
Dalia (2017) Case report 61 years/M Perforation Abdominal CT scan Chemotherapy Died 4
Cabrera (2016) Case report 71 years/M No Abdominal CT scan, None Recovered 4
endoscopy
Zhu (2015) Case report 62 years/M No Abdominal CT scan None Recovered 4
43 years/M No Abdominal CT scan None Recovered 4
73 years/M No Abdominal CT scan None Recovered 4
Yamasaki (2016) Case report 42 years/F Ascites, metastasis Abdominal CT scan Chemotherapy Died 4
Habib (2009) Case report 38 years/M No Abdominal CT scan None Recovered 4
Damiano (2011) Case report 88 years/F Metastasis, Abdominal CT scan, Chemotherapy Died 4
obstruction colonoscopy
Hee (2012) Case report 41 years/M No Abdominal CT scan None Recovered 4
Kenoki (2015) Case report 20 years/M No Abdominal CT scan None Recovered 4
Kamal (2016) Case report 20 years /M No Abdominal CT scan None Recovered 4

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In these articles, patients complained of
abdominal mass (45.5%) and abdominal
pain (45.5%). Preoperative diagnosis
established using CT scan (91%) and
abdominal ultrasound (9%). On
histopathological findings, seven articles
(63.6%) of duplication of intestine
transformed into adenocarcinoma, and
two articles were non–malignant
(18.2%), and two other articles
transformed into transitional cell
carcinoma and GIST.
Out of five articles, the preventive
surgery was proceeded in four articles
with the outcome was recovered in 80%
subjects and died preceded by recurrent
in 20% subject. The conservative
proceeded in six other articles; four
articles reported mortality of 50%,
recovery of 33%, and recurrent of 67%.
Discussion
The articles subjected to analysis in this
study were case reports and those with
level of evidence IV. Thus, to decide
whether preventive or conservative
method when dealing with intestinal
duplication based on these studies
requiring a very careful judgement. It is
reasonable as intestinal duplication is an
infrequent case.
The malignant changes that developed
from intestinal duplication may be found
in vary, namely adenocarcinoma (the
most common type), squamous cell
carcinoma and transitional cell
2,3
carcinoma. These changes were
reported on seven articles, which is 63%
developed to adenocarcinoma. Even
though the incidence is infrequent,
development of malignancy as much as
63% is quite high and should be of one
consideration.
Putranto et al
There are two surgical interventions in
the management of intestinal duplication
currently applied, namely conservative–
and preventive surgery. Conservative
surgery defined as resection of the
duplicated and normal intestine to a
certain extent, in accordance with the site
of duplicated part. While as preventive
surgery referred to a surgical resection in
accordance with oncologic principles,
which is radical, including lymph nodes
dissection. The outcome is found in vary,
namely recovered with no complication
and back to normal activity or
postoperative mortality.
Both of treatment requiring critical
consideration with individual approach.
First, it should be realized that in most
articles, the intestinal duplication was
suspected preoperatively. In this case,
any diagnostic tools to sharpen the
diagnosis is justified. However, in
emergency setting, the priority in the
management is to solve the problem
encountered. Intussusception, which is
an emergency case of strangulated bowel
need an emergency surgery that
addressed to prevent intestinal necrosis,
where sharpening the diagnosis with
abdominal CT is not justified in this
setting. In this setting, ultrasound may be
of some preference as it is practical and
may shows the features of
intussusceptions, that surgeons can do.
When dealing with intussusceptions in
adults, this unusual entity should be
carefully addressed. Careful assessment
of possible cause is important. However,
it is premature to decide preventive is
better than conservative based on these
studies since there is many factors to be
considered. Should the abdominal
assessment represent the stigmata
suggesting malignancy such as nodules,
lymph node enlargement etc. in addition
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to history of malignancy symptoms, then
surgical resection with oncological
approach is justified. In contrast,
conservative treatment should be
adequately followed up for possible
unexpected development of malignant
degeneration.
Conclusion
Case reports in the study showed that
63% of intestinal duplication were
developed to malignancy, requiring
preventive surgery (level of evidence
IV). However, this should be judge with
a careful clinical assessment.
Disclosure
Authors declare no conflict of interest
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