Clinical Case Report Medicine ®

OPEN

A huge completely isolated duplication cyst

complicated by torsion and lined by 3 different
mucosal epithelial components in an adult
A case report
∗
Ai Xiao-Ming, MD , Lu Jin-Jing, MM, Ho Li-Chen, MM, Han Lu-Lu, MM, Yue Xiong, MM,
Zhang Hong-Hai, MM, Yang Nian-Yin, MD

Abstract
Rationale: Intestinal or enteric duplication (ED) does exit as a rare congenital malformation of the gastrointestinal system clinically. It
is a separate entity, but can be communicated with the gastrointestinal tract. It is characterized by a well-developed muscular wall
and lumen endowed with ectopic mucosa, simulating a portion of normal bowel. A completely isolated duplication cyst (CIDC) refers
to an extremely uncommon variant of ED, which is secluded from the alimentary tract and possesses its own exclusive blood supply.
Surgical procedure is the treatment of choice, because most often, a definitive diagnosis can only be confirmed intraoperatively.
Patient concerns: A 20-year-old male patient presented with a 10-day history of intermittent episodes of abdominal pain. The
pain evolved from dull into progressive and intolerable, accompanied by vomiting, nausea, and abdominal distention.
Diagnoses: Closed-loop small-bowel obstruction with volvulus.
Interventions: The patient underwent an emergency exploratory laparotomy.
Outcomes: A huge CIDC was observed upon operation, which was affixed to the mesentery with only a narrow base, just like a
pedicle; 720° counterclockwise twisting around its base was definitely noted, provoking the compromised blood supply. Complete
excision of the cyst was performed along its base safely without violating the intestinal tract. Furthermore, the ectopic mucosa of the
cyst exhibited 3 different epithelial lining components histopathologically.
Lessons: Clinicians should be aware of the possibility of the existence of a duplication and raise a high index of suspicion in case of
equivocal diagnosis, particularly in adult population. A low threshold for surgical management should be recommended in order to
prevent lethal outcomes.
Abbreviations: CIDC = completely isolated duplication cyst, CT = computed tomography, ED = enteric duplication, EDC =
enteric duplication cyst, US = ultrasonography.
Keywords: completely isolated duplication cyst, intestinal or enteric duplication, mucosal lining, torsion

1. Introduction time been perplexing pediatric surgeons. It is really a challenging

task to make a clinical diagnosis mainly due to its rarity and
As a potentially life-threatening disease, an enteric duplication
nonspecific presentations, unless complications ensue unexpect-
(ED) or intestinal or enteric duplication cyst (EDC) has all the
edly. In addition, it affords little opportunity to elucidate its
mysterious nature and characteristics.
Editor: N/A. An EDC is an unusual congenital deformity of the alimentary
Ai Xiao-Ming and Lu Jin-Jing have contributed equally to this article. system, which is a separate entity invested with a cystic appearance,
Informed written consent has been obtained from the patient for the publication but at the same time is in intimate contact or communication with
of this case report. the alimentary tract.[1] This entity consists of a wide variety of
This case report was reviewed and approved by the ethic committee of BenQ cystic lesions originating from different sites alongside the digestive
Medical Center.
tract between the mouth and the anus in conjunction with their
The authors have no funding and conflicts of interest to disclose. neighboring organs, with a predilection for the small bowel,
Department of General Surgery, BenQ Medical Center, The Affiliated BenQ especially the ileum.[2,3] It takes place most commonly in infants
Hospital of Nanjing Medical University, Nanjing, Jiangsu, China.
∗
and children, with an estimated incidence of approximately 1 in
Correspondence: Ai Xiao-Ming, BenQ Medical Center, Nanjing, Jiangsu, China 4500 births,[4] but is exceedingly uncommon in adults.
(e-mail: aixiaoming0503@126.com).
A completely isolated duplication cyst (CIDC) refers to an
Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc.
extremely rare variant of EDC, which does not contact with or is
This is an open access article distributed under the terms of the Creative
Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC- secluded from the alimentary tract and possesses its own exclusive
ND), where it is permissible to download and share the work provided it is blood supply.[5] A CIDC occurs much more scarcely, with a
properly cited. The work cannot be changed in any way or used commercially prevalence of about 1 in 10,000 live births,[6] and to the best of our
without permission from the journal. knowledge, there have been only 10 cases of CIDC in adults ever
Medicine (2018) 97:44(e13005) published in English literature,[5,7–15] among which 2 experienced
Received: 3 July 2018 / Accepted: 5 October 2018 malignant degeneration.[12,14] Herein, we reported a case of a
http://dx.doi.org/10.1097/MD.0000000000013005 20-year-old male patient who presented with a huge CIDC, which

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Xiao-Ming et al. Medicine (2018) 97:44 Medicine

served as the lead point for the cystic torsion, adding up to the proximal to the ileocecal junction and assumed a smooth surface.
increased difficulty for diagnosis and rendering the mandatory It was apparently distended and ischemic but without obvious
emergency laparotomy. Interestingly, the ectopic mucosa of the gangrene or necrosis. On first impressions, this cyst appeared to
cyst was lined with 3 different epithelial components histopatho- be a swollen small bowel loop (Fig. 2). Whereas, it was neither
logically. To date, such a case has never been reported yet. connected nor adhesive to the adjacent bowel. In fact, this object
was only affixed to the mesentery with a narrow base, just like a
pedicle constricting its feeding vessel, and it was in no small part
2. Case report
mobile, blind and secluded from the bowel lumen. Furthermore,
A 20-year-old male patient was submitted to the emergency with a 720° counterclockwise twisting around its base was definitely
10-day history of intermittent episodes of abdominal pain without noted, which provoked the compromised blood supply. There
any obvious predisposing cause. The pain seemed dull at first, but was approximately 50 mL turbidly purulent ascites additionally.
gained progressive and intolerable gradually, which was then Subsequently, following a clockwise untwisting, the cyst was
accompanied by vomiting, nausea, along with abdominal distention. excised along its base safely without violating the intestinal tract.
The discomfort had not settled with conservative treatment. The Grossly, the resected specimen measured 70 cm in length and 8
patient was otherwise healthy without such an onset ever before. cm in maximal diameter, which was unilocular and contained
On examination, he appeared anguished, but still maintained massive hemorrhagic fluid intermixed with clots. Histopathology
normal vital signs. His abdominal examination revealed (Fig. 3) illustrated that the cystic wall was uniquely composed of a
tenderness over the lower abdomen, with no rigidity, rebound heterotopic mucosa combined with intact underlying well-
tenderness, or voluntary guarding. No palpable mass was defined smooth muscle layers, resembling a part of normal
palpated and the bowel sound was as usual. The laboratory bowel tract. All these findings confirmed a final diagnosis of an
investigations were only remarkable for an elevated white blood ED consistent with congenital malformation. A variety of
cell count of 17.28  109/L, with NEUT% 85.5%. The plain mucosal lining types were displayed throughout the lumen:
abdominal X-rays provided no special findings. The ultrasonog- segmented squamous epithelium, focal gastric glandular mucosa
raphy (US) visualized significantly dilated bowel loops together of foveolar and oxyntic type, in addition to patchy intestinal
with a small amount of abdominal fluid. The contrast-enhanced metaplasia. The postoperative recovery course was uneventful
computed tomography (CT) scan (Fig. 1) was subsequently without any complications or events. The patient was discharged
performed and demonstrated multiple evidently dilated cystic- on the 7th postoperative day and still remained well over the
like small-bowel loops with thickened walls, communicating with routine follow-up.
each other. There was twisting of mesenteric vessels around each
other suggesting the whirlpool sign, apart from a mild portion of
3. Discussion
ascites. Taken together, features on the CT scan were highly
indicative of closed-loop small-bowel obstruction with volvulus An ED is quite infrequently seen in an adult. Reportedly, it
to be the likely cause. Therefore, the patient was admitted to the usually occurs during the neonatal or childhood period, mostly
department of general surgery immediately. After his endorse- before the age of 2,[16] and is slightly predominant in males.[16]
ment of the informed consent, he underwent an emergency Although this extraordinary anomaly is an independent entity,
exploratory laparotomy. most often it is adherent to or contiguous with the main intestinal
At surgery, the intra-abdominal exploration demonstrated that tract. However, it is constantly variable in location and anatomy.
a huge cystic-like object was located in the ileal mesentery 45 cm It can be sited on either the mesenteric or the antimesenteric side

Figure 1. Features of the duplication on computed tomography scan. (A) A demonstration of a well-marginated cyst mistaken initially as a distended bowel loop
(arrow), which was definitely an intestinal duplication verified intraoperatively and retrospectively, full of hemorrhagic fluid. (B) A description of twisting of mesenteric
vessels around each other at the base of the cyst (arrow), suggestive of the whirlpool sign.

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Figure 2. Intraoperative findings of the detorsioned duplication cyst. (A) The distended, ischemic, and separate cyst, measuring 70 cm in length and 8 cm in
maximal diameter; (B) the attachment of the cyst to the ileal mesentery with a pedicle, without any communication with bowel.

of the bowel wall, with a mesenteric preponderance,[1] and its multilobulated.[18] Frequently, a duplication is mobile, and can
length ranges from few centimeters to whole length of the small be observed as a palpable mass in 50% of cases.[19] Rarely can an
intestine.[17] Most of the time, it is single, while concurrent ED have its own autonomous blood supply independent from the
multiple cysts are exposed in 10% to 15% of cases. It appears neighbouring bowel tract; conversely, it shares the common
unilocular mostly, but sometimes could be multilocular, or even vessel system with the mesentery.[20] Even some additional

Figure 3. Histopathological configuration of the duplication. (A) The cystic wall containing an ectopic mucosa with a variety of epithelia linings covering the cystic
cavity, and overlying the smooth muscle layer (hematoxylin and eosin [HE], 40). (B) An illustration of the focal area of mucosal transition between squamous and
glandular epithelia (HE, 100). (C) A depiction of the small portion of gastric type mucosa with foveolar and oxyntic glands easily appreciated, secreting mucous
substances (HE, 100). (D) A representative section of formation of the intestinal metaplasia, showing a sheet of enteric type mucosa rich in goblet cells and Paneth
cells, which were infiltrated with myriads of erythrocytes (HE, 100).

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associated anomalies have been encountered in 50% of patients, on.[32] Nevertheless, none of them have been persuasive enough
the majority being vertebral defects.[1,16] An ED can also be alone. Thence, the precise mechanism still remains poorly
correlated with malrotation.[21] Our current patient was not understood.
found to have other anomalies or malrotation, with the Regarding its symptoms it is particularly difficult to distinguish
duplication located in the mesentery separately. an ED from other intra-abdominal cystic etiologies.[8,19] Besides,
By definition, an ED or EDC is characterized by a well- it is quite confused with a Meckel’s diverticulum when locating
developed muscular wall along with a lumen lined by ectopic on the antimesenteric side. Its manifestations may be vague, and
mucosa,[11] which bears a striking resemblance to a portion of the lack of specificity, mimicking other diseases.[3,33] Although it can
normal bowel tract. It is endowed with the similar layered present with acute abdomen during the neonatal or childhood
muscles with the normal bowel wall, and frequently even shares a period, even progressing to unexpected death, it could also
common muscular wall. The ectopic mucosa is typically lined by remain asymptomatic until at school age or adulthood.[3]
gastrointestinal-type epithelium ranging from gastric to rectal Nonetheless, the vast majority of cases would develop symptoms
component,[22] which is usually compatible with the adjacent in the first year of life.[34] Of importance, most often, only can
bowel, while different epithelial constituent other than gastroin- enough attention be paid to the very cases due to inadvertent
testinal origin has been sometimes identified, commonly being manifestation of complications, including intestinal obstruction,
respiratory epithelium.[22] Normally, a single epithelial lining is perforation or cystic rupture, volvulus or cystic torsion, infection,
present, the majority being gastric,[2] although a combination of or malignancy.[4]
multiple types of epithelia coexisting in the same duplication has Numerous imaging modalities may help in diagnosis of an ED.
occasionally been disclosed.[1,23] Besides, infrequently, tissues US plays a crucial role in the evaluation of a duplication, and has
like an annular or aberrant pancreas are also observed,[24] but the capability of showing the characteristic double wall signs or
cartilage is never shown within the wall.[25] In our case, gut signature signs.[29] Many thanks to its improved accuracy,
squamous epithelium, which is of disparate origin and seldom prenatal US has identified a good number of cases of duplication
emerges in the normal abdominal cavity, in combination with 2 in utero.[1] However, this very sign could sometimes be
other gastrointestinal-type epithelial compartments, uniquely misleading,[34] which is also observed in cysts of other geneses
comprised the mucosal lining. (false positive),[35] such as mesenteric cyst, Meckel’s diverticu-
Functional secretion of the ectopic mucosa of an ED tends to lum, ovarian cysts, and cystic teratoma, and so on. Thus far, only
produce a clear mucoid fluid in the hollow lumen,[20] leading to 20% to 30% of lesions with prenatal US have been detected,[4]
distention of the cyst. In terms of ectopic gastric mucosa, and an overall sensitivity of merely 55% has been obtained
excessive acid secretion may give rise to ulceration, erosion, pertaining to the diagnosis of ED actually.[36] Magnetic
bleeding, or perforation.[26] By contrast, heterotopic pancreatic resonance imaging, which is recommended to prenatal utility
tissue could trigger increased pancreatic enzymes contained in the as well, is as diagnostic as US, but unable to provide any more
fluid,[27] even hypoglycemia.[26] An ED uncovered in the neonates useful information.[20] Barium studies may be helpful if the
is benign, but can develop malignant transformation due to the duplication is tubular,[1] communicating with the bowel lumen,
persistent presence and stimulation of ectopic mucosa, which is and a gastrointestinal endoscopy might be equally feasible upon
solely seen in adults[4,11] and in the form of adenocarcinoma.[28] such an occasion. In the context of gastrointestinal bleeding, Tc-
There was no evidence of dysplasia or malignancy in our case, 99m pertechnetate radionuclide scan is liable to discern the
and the mucosal secretion potentially contributed to its heterotopic gastric mucosa, which aids in the discrimination of
presentations and complications. Meckel’s diverticulum from a duplication when locating on the
Generally speaking, there are 2 chief categories of duplication antimesenteric border of the small bowel, with an up to 75%
according to morphology[20,29]: one is tubular, while the other sensitivity.[37] Nonetheless, it is worth noting that heterotopic
cystic or spherical. The tubular communicates with the bowel gastric mucosa does emerge in only 20% to 30% of duplication
lumen, whereas the cystic representing the vast majority of cases, cysts,[38] which dramatically limits the detectable usefulness of
does not. The duplication in our case was reasonably cystic, with isotope studies.[39] Enhanced CT scan is a more common and
no communication. Meanwhile, on the basis of the vascular desirable imaging tool, offering further characteristics of the
pattern, Li et al[30] have classified small EDs as type 1 or parallel lesion along with delineation of the surrounding structures,[33]
type (74.6%) and type 2 or intramesenteric type (24.4%). The which most likely indicates the necessity of immediate management.
duplication in our case belonged to type 1 logically: the cyst In our patient, US failed to reflect the duplication, with merely a
resided on one of the leaves of the mesentery and its feeding artery perception of puffy bowel loops, which was principally ascribed
was separate from that of the bowel. The most important to lack of suspicion, little familiarization with its appearance, as
implication of this classification may possibly lie in its well as obstacles created by trapped gas in the enlarged bowel
determination of complete excision without disturbing the loops. But we are convinced that such US findings of dilated
continuity of the bowel, just as depicted in our patient. bowel loops would be highly suspicious of an ED or intra-
A CIDC was present in our patient, which had a dedicated abdominal cyst should it take place in a neonate or child. In light
vascular peduncle, in keeping with description in some other of the noteworthy information of CT scan indicative of bowel
literature.[11] It just hung free on the mesentery, which made it obstruction and volvulus, we determined to take prompt
susceptible to twisting around the peduncle, serving as the lead measures for surgical treatment, thus potentiating the final
point for torsion. Plausibly, there has already raised a speculation diagnosis and satisfactory prognosis. Nevertheless, on review of
that an ED might predispose the patient to volvulus.[31] the CT scan and consultation with the radiologists, we are
As to the pathogenesis, it is probably related to embryonic confident that the markedly enlarged bowel loop (Fig. 1A) was
development disorders. Several major theories have been definitely an ED, in accordance with the patterns described in the
hypothesized,[29] to explain the development of duplication literature.[29,34]
arising in different sites, such as vacuolization, caudal twinning, Unfortunately, a definitive diagnosis in most cases is only
diverticularization, as well as split notochord theories and so confirmed incidentally, especially intraoperatively,[1] irrespective

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of the assist of imaging methods, just as shown in our case. Once [6] Simsek A, Zeybek N, Yagci G, et al. Enteric and rectal duplications and
duplication cysts in the adult. ANZ J Surg 2005;75:174–6.
torsion or volvulus occurs, it can progress to strangulation or
[7] Blank G, Königsrainer A, Sipos B, et al. Adenocarcinoma arising in a
necrosis abruptly, thus contributing to the deteriorated risk of cystic duplication of the small bowel: case report and review of literature.
morbidity and mortality. Hence, a low threshold for surgical World J Surg Oncol 2012;10:1–4.
management should be recommended in cases of suspected [8] Kim SK, Lim HK, Lee SJ, et al. Completely isolated enteric duplication
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[9] Nichols KC, Pollema T, Moncure M. Laparoscopically excised
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[11] Park JY, Her KH, Kim BS, et al. A completely isolated intestinal
the adjacent bowel is mandatory on account of the intimate
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resection should be discouraged as far as possible, in avoidance of [12] Shin SY, Cho MY, Ryu H, et al. Adenocarcinoma originating from a
short bowel syndrome, and upon such a setting, some alternative completely isolated duplication cyst of the mesentery in an adult. Intest
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4. Conclusion completely isolated infected ileal duplication cyst. S Afr J Surg
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[15] Pant N, Grover JK, Madan NK, et al. Completely isolated enteric
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[17] Ekbote G, Pokharkar AB, Moon P. A rare case of perforated tubular
population. Once taking the suspicion into consideration, just as
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depicted in our patient, either an exploratory laparotomy or, 20.
whenever possible, laparoscopy should be designated in order to [18] Momosaka D, Ushijima Y, Nishie A, et al. A retroperitoneal isolated
verify the diagnosis, to facilitate a timely treatment as well as to enteric duplication cyst mimicking a teratoma: a case report and
eliminate progression of severe complications. literature review. Case Rep Radiol 2016;2016:1–5.
[19] Tiwari C, Shah H, Waghmare M, et al. Cysts of gastrointestinal origin in
children: varied presentation. Pediatr Gastroenterol Hepatol Nutr
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Author contributions [20] Liaqat N, Latif T, Khan FA, et al. Enteric duplication in children: a case
Ai Xiao-Ming, Lu Jin-Jing, and Ho Li-Chen conceived the series. Afr J Paediatr Surg 2014;11:211–4.
[21] Srivastava P, Gangopadhyay AN, Kumar V, et al. Noncommunicating
research idea, designed this study, as well as drafted the isolated enteric duplication cyst in childhood. J Pediatr Surg 2009;44:e9–
manuscript. Ai Xiao-Ming, Han Lu-Lu, and Yue Xiong 10.
completed the clinical data collection and performed histological [22] Ladd MR, Garcia AV, Allison DB, et al. Intraluminal Meckel’s
examination. Yang Nian-Yin and Zhang Hong-Hai interpreted duplication cyst causing bowel obstruction in an infant: a role for
the clinical data and revised the manuscript. All the authors read laparotomy. Case Rep Pediatr 2016;2016:4717403.
[23] Shah A, Du J, Sun Y, et al. Dynamic change of intestinal duplication in an
and approved the final manuscript. adult patient: a case report and literature review. Case Rep Med
Conceptualization: Ai Xiao-Ming, Ho Li-Chen, Han Lu-Lu. 2012;2012:297585.
Data curation: Ai Xiao-Ming, Lu Jin-Jing, Yue Xiong. [24] Ogino H, Ochiai T, Nakamura N, et al. Duplication cyst of the small
Investigation: Ai Xiao-Ming, Lu Jin-Jing, Yue Xiong. intestine found by double-balloon endoscopy: a case report. World J
Gastroenterol 2008;14:3924–6.
Supervision: Yang Nian-Yin. [25] Singhal V, Shenoy RD, Kamath N, et al. Mediastinal enteric cyst in a
Validation: Zhang Hong-Hai, Yang Nian-Yin. neonate. J Clin Neonatol 2012;1:149–51.
Writing – original draft: Ai Xiao-Ming, Ho Li-Chen, Han Lu-Lu. [26] Mansi M, Mahajan N, Mahana S, et al. Aberrant pancreatic tissue in a
Writing – review & editing: Zhang Hong-Hai, Yang Nian-Yin. mediastinal enteric duplication cyst: a rarity with review of literature.
Case Rep Gastrointest Med 2017;2017:7294896.
Ai Xiaoming orcid: 0000-0002-7743-9631.
[27] Sultan M, Karanovic D, Chalhoub W, et al. Gastric duplication cyst with
elevated amylase: an unusual presentation mimicking pancreatic cystic
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