CASE REPORT – OPEN ACCESS

International Journal of Surgery Case Reports 42 (2018) 295–298

Contents lists available at ScienceDirect

International Journal of Surgery Case Reports

journal homepage: www.casereports.com

Ciliated cyst of the gallbladder: A new case and literature review

Triki Wissem ∗ , Baraket Oussema, Itami Ahmed, Baccar Abdelmajid, Marzouk Ines,
Bouchoucha Sami
Tunis El Manar University, Department of General Surgery, Habib Bougatfa Hospital, Bizerte, Tunisia

a r t i c l e i n f o a b s t r a c t

Article history: INTRODUCTION: The ciliated cyst is a rare anomaly that develops from the anterior primitive intestine.
Received 7 November 2017 Its localization is essentially supra diaphragmatic. It’s localization in the gallbladder is very uncommon.
Received in revised form CASE REPORT: We report the first case in Tunisia of a ciliated cyst of the gallbladder in a 34 years old
29 November 2017
woman who was operated for a gallbladder stone and in whom the discovery of a cystic mass attached
Accepted 1 December 2017
to the neck of the gallbladder was preoperatively. The pathologic study did conclude to a ciliated cyst.
Available online 21 December 2017
DISCUSSION: The ciliated cyst of the gallbladder is a benign congenital lesion that develops from the
anterior primitive intestine. The most frequent clinical symptom is abdominal pain and the median age
Keywords:
Ciliated foregut cyst is 45 years old. The most frequent location is the neck. This pathology affects women more than men.
Gallbladder cyst Abdominal ultrasound is not very specific exam and describes the ciliated cyst as a cystic lesion often
Anomaly anechogenic and sometimes hyperechogenic. The CT-scan as well as the abdominal MRI are very helpful
of the diagnosis. The positif diagnosis of ciliated cyst is histological.
CONCLUSION: The recommended current treatment for this rare pathology is surgery and it consists of
a celioscopic cholecystectomie. The place of conservatory treatment hasn’t been established due to the
rarity of described case and the possibility of degeneration.
© 2017 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open
access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

1. Introduction 2. Observation

The ciliated cysts is a rare congenital lesion developed from the
primitive anterior intestine who is the origin of the oropharynx,
the bronchio-pulmonairytractus, the esophagus, the stomach, the
duodenum, the liver, bile ducts and the pancreas. The ciliated cyst is
essentially supra-diaphragmatic and located in the bronchial tree,
the esophagus, the mediastinum and sublingual. When the cyst
is infra diaphragmatic, it’s usually located in the liver especially
in medial segments (IV,V,VIII). In can be located in the pancreas
as well. It’s location in the gallbladder is exceptional. Therefore,
few cases were published. To our knowledge, only 15 cases were
described in literature. We report the first case diagnosed in Tunisia
and the 16th in the world with a review of literature. This work has
been reported in line with the SCARE criteria [1].
A 34 years old patient with no medical history was admitted for
an upper right quadrant pain. The clinical examination found right
upper quadrant tenderness. Blood test showed no anomalies and
the abdominal ultrasound showed a gallbladder stone with thin
intra and extra hepatic bile and hilar adenopathy. The abdominal
computed tomography reveled unilocular cystic lesion attached to
the neck of the gallbladder (Fig. 1). The patient was operated via
a right subcostal incision. The paraopeartively exploration found a
gallbladder with thin walls and a 2 cm round cystic mass attached
to the neck of the gallbladder. She had a cholecystectomy and the
follow ups were simple. The anatomopathologic study found a cys-
tic lesion with thin walls covered with ciliated epithelium which
lays on a connective tissue that contains smooth muscle fibers
(Figs. 2 and 3). The content of the cyst was a mucoid liquid and
there was no communication between the cyst and the gallbladder
lumen. There was no sign of degeneration.

3. Discussion

∗ Corresponding author at: Departement of Surgery, Habib Bougatfa Hospital,
7000 Bizerte.
E-mail addresses: wissem triki@yahoo.com (T. Wissem), oubaraket@gmail.com
(B. Oussema), ahmed.itaimi@hotmail.com (I. Ahmed),
abdelmajidbaccar@gmail.com (B. Abdelmajid), marzouk.ines@gmail.com (M. Ines),
sami.bouchoucha@yahoo.com (B. Sami).
Cystic lesions of the gallbladder are rare and they can origi-
nally congenital, acquired or neoplastic [2]. The ciliated cyst of the
gallbladder is a benign congenital lesion that develops from the
anterior primitive intestine. Cysts are essentially supradiaphrag-
matic in the bronchial tree, the esophagus, the mediastinum and

https://doi.org/10.1016/j.ijscr.2017.12.024
2210-2612/© 2017 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license (http://
creativecommons.org/licenses/by-nc-nd/4.0/).
 CASE REPORT – OPEN ACCESS
296 T. Wissem et al. / International Journal of Surgery Case Reports 42 (2018) 295–298
Fig. 1. A unilocular cystic lesion attached to the neck of the gallbladder.
Fig. 2. the cyst was lined by pseudostratified ciliated columnar epithelium.
Fig. 3. A low power view of the complex columnar cellined gallbladder mucosa at
the top of the field, and at the lower edge a single layer of ciliated epithelium.
sublingual. Under the diaphragm, the ciliated cysts are in the liver
and develop from intrahepatic bile ducts. Its location in the gall-
bladder is exceptional.
There is many hypotheses that describes the pathogenesis of
ciliated cysts [3]. Kakitsubata described in 1995 the first case of
gallbladder cyst who was covered with cylindrical ciliated epithe-
lium and he named it “Epithelial cyst of the gallbladder” [4]. In
2000, another case was reported by Nam under the title of “Cil-
iated foregut cyst of the gallbladder” [5]. Afterward, only twelve
cases were published.
Tables 1 and 2 present a resume of literature review that per-
mits to deduce the epidemiological and pathologic characteristics
related to this entity.
This pathology affects women more than men (10 women to
5 men) with a sex ratio of 2. The median age is 45 years old
with extreme ages of 9–75 years old. Only one case of an infant
was noted. The median size of the cyst is 2.5 cm with extremes of
0.7–3.5 cm. The most frequent location is the neck, it was observed
in 6 cases followed by the body with 4 observed cases. In three
patients the cyst was located in the fundus. The location wasn’t
mentioned in 2 cases. The most frequent clinical symptom is
abdominal pain which was noted in 6 patients. In 5 cases, the
discovery of the gallbladder cyst was incidental. On patient had
cholecystitis and another one had epigastric pain and vomiting.
In two observations, clinical symptoms weren’t mentioned. All
patients underwent radiologic exams preoperatively. In two thirds
of cases (11 cases), ultrasound revealed a cystic lesion. In one case,
ultarsound revealed gallbladder stones and in another an acute
cholecystitis. The aspect of diverticula or duplication of the gall-
bladder was observed in one case. In our patient, the gallbladder
lesion was mistakenly interpreted as an adenopathy while CT-scan
confirmed the presence of an aspect of a cyst. In 100% of cases, the
cyst was unilocular. In the majority of cases, the cyst content was
mucoid (9 patients) while gelatinous in 2 patients, dark brown in 2
others and milky in one patient.
The positif diagnosis of ciliated cyst is histological. On an anato-
mopathogic level, the cystic lesion is caraterized by a thin wall
covered with a cylindrical ciliated mucus-secreting epithelium
which lays on connective tissue containing smooth muscular fibers.
There is now communication between the cyst and the gallbladder
lumen.
Concerning imaging techniques, abdominal ultrasound is the
fist most sought exam in exploring hepatic and biliairy diseases.
Although it is not very specific, it describes the ciliated cyst as a
 CASE REPORT – OPEN ACCESS
T. Wissem et al. / International Journal of Surgery Case Reports 42 (2018) 295–298 297

Table 1
Reported case of cyst in gallbladder.

Author Sex Age Clinic Imaging Location Size (cm)

Kakisubata [4] M 71 nil Anechogenic cystic lesion of the body unspecified

gallbladder
Benlolo [9] F 75 unspecified unspecified fundus 3
Nam [5] F 36 nil Anechogenic cystic lesion and fundus 1.5
hypodense in CT-scan
Chatelain [10] F 75 unspecified Echogenic cystic lesion unspecified 3.3
Hirono [11] F 43 nil Cystic mass neck 2.5
Muraoka [12] F 36 nil Cystic lesion with hyperechogenic body 2.4
focal points, tumoral aspect in
CT-scan
Bulut [13] F 41 Right upper quadrant pain Benign cystic lesion neck 3.5
Kim [14] M 63 nil Cystic mass unspecified 2
Tuncyurek [6] F 42 Right upper quadrant pain Anechogenic cystic lesions with body 0.7
hyperechogenic focal points
Angot 2013 M 45 Right upper quadrant pain with Cholecystitis with gallbladder body 3
fever sludge
Giacoustidis [15] F 29 Epigastric pain with diarrhea and Cystic mass adjacent to the neck 3
vomiting gallbladder et al a cholangio MRIa
pouch connected to the cystic duct
containing debris
Lee [16] M 61 Right upper quadrant pain Gallbalder stone fundus 2.7
Agarwal 2016 [17] M 9 Right upper quadrant pain with Diverticula or duplication of the neck 3
vomiting gallbladder
Hwang [18] F 39 Right upper quadrant pain Cystic lesion neck 3.5
Eun Han [19] F 20 Right upper quadrant pain Cystic lesion attached to the neck neck 1.6
of the gallbladder
Our case F 34 Right upper quadrant pain Gallbladder stone neck 3

Table 2
Pathologic characteristics of ciliated cysts.

Author Locularity Cyst content Cyst wall Malignity

Kakisubata [4] Unspecified Unspecified Unspecified Unspecified

Benlolo [9] Unilocular Dark brown Unspecified Unspecified
Nam [5] Unilocular Mucoid Thin No
Chatelain [10] Unspecified Mucoid Unspecified Unspecified
Hirono [11] Unilocular Dark brown Unspecified Unspecified
Muraoka [12] Unilocular Mucoid Thin Unspecified
Bulut [13] Unilocular Mucoid Unspecified Unspecified
Kim [14] unspecified Milky Unspecified Unspecified
Tuncyurek [6] unilocular Dense Mucoid Unspecified No
Angot 2013 unilocular Mucoid Thin No
Giacoustidis [15] unilocular Gelatinous grey Unspecified Unspecified
Lee [16] unilocular gelatinous Thin No
Agarwal [17] unilocular Mucoid Thin No
Hwang [18] unilocular Mucoid Thin No
Eun Han [19] unilocularunilocular Mucoid Unspecified Unspecified
Our case unilocular mucoid Thin No

cystic lesion often anechogenic and sometimes hyperechogenic. In 4. Conclusion

our patient, the abdominal ultrasound didn’t establish the diagno-
sis. When the lesion is hyperechogenic the diagnosis of a neoplastic
origine can be evoqued [6] especially when the cysts size is exceeds
1 cm. In this case the CT-scan as well as the abdominal MRI are very
helpful of the diagnosis. The ciliated cyst is often observed in hyper-
signal T1 and T2 and certain cysts can appear in hyposignal T1. In
fact, the contrast depends in parts on the viscosity of the cyst con-
tent and in other part on the presence or absence of calcium and
cholesterol crystals.
Treatment is based on the cholecystectomy [7]. Although, Tun-
cyurek proposed a close surveillance when the cyst is silent [6]. The
systematic cholecystectomy seems the most wise attitude since
the distinction between benign and malignant lesion is difficult
on imaging techniques and all the more that cases of malpigien
metaplasia and epidermoid carcinoma that occurred on hepatic
ciliated cysts were observed [8]. Even though no case of malignat
transformation of the ciliated gallbladder cyst was described, it’s
convenient to look thoroughly into the resection specimen.
The ciliated cyst of the gallbladder is a congenital rare cyst.
The preoperative diagnosis is still difficult to establish despite
the progress of imaging techniques. The positive diagnosis is
established on anatomopathogic data. The recommended current
treatment is surgery and it consists of a celioscopiccholecystec-
tomie. The place of conservatory treatment hasn’t been established
due to the rarity of described case and the possibility of degenera-
tion.
Conflicts of interest
There aren’t any conflicts of interest for all authors.
Funding
We have no sources of funding.
 CASE REPORT – OPEN ACCESS
298 T. Wissem et al. / International Journal of Surgery Case Reports 42 (2018) 295–298

Ethical approval [3] R.J.R. Cureton, J.F. Newcombe, Multilocular cyst of the gallbladder, Br. J. Surg.
48 (1961) 577–580.
[4] Y. Kakitsubata, S. Kakitsubata, K. Marutsuka, K. Watanabe, Epithelial cyst of
Ethical approval has been exempted by our institution. the gallbladder demonstrated by ultrasonography: case report, Radiat. Med.
13 (1995) 309–310.
Consent [5] E.S. Nam, H.I. Lee, D.H. Kim, et al., Ciliated foregut cyst of the gallbladder: a
case report and review of the literature, Pathol. Int. 50 (2000) 427–430.
[6] Ö. Tunçyürek, D. Nart, B. Yaman, E. Buyukcoban, A ciliated foregutcyst in a
Our study is a case report. The consentement of the patient has gallbladder: the smallest recorded, Jpn. J. Radiol. 31 (2013) 412–418.
been obtained. [7] T. Oida, K. Mimatsu, A. Kawasaki, et al., Laparoscopic excision for an enlarged
ciliated hepatic foregut cyst as a minimally invasive procedure, J.
Laparoendosc. Adv. Surg. Tech. A 19 (2) (2009) 203–206.
Author contribution [8] N. Ben Mena, S. Zalinski, M. Svrcek, et al., Ciliated hepatic foregut cyst with
extensive squamous metaplasia: report of a case, Virchows Arch. 449 (2006)
730–733.
Wissem Triki: development of the idea and writing of the article. [9] D. Benlolo, V. Vilgrain, B. Terris, M. Zins, J. Belghiti, Y. Menu, Imaging of
Oussema Baraket and Ahmed Itami: bibliographic search Ines ciliated hepatic or biliary cysts: four cases, Gastroenterol. Clin. Biol. 20 (1996)
Marzouk: provision and interpretation of radiological images. 497–501.
[10] D. Chatelain, B. Chailley-Heu, B. Terris, G. Molas, A.L. Cae, V. Vilgrain, et al., The
Abdelmajid Baccar: provision and interpretation of anato-
ciliated hepatic foregut cyst, an unusual bronchiolar foregut malformation: a
mopathologic images. histological, histochemical and immunohistochemical study of 7 cases, Hum.
Sami Bouchoucha: final revision. Pathol. 31 (2000) 241–246.
[11] S. Hirono, H. Tanimura, S. Yokoyama, K. Uchiyama, M. Tani, H. Onishi, et al.,
Clinical features of ciliated foregut cyst of the gall bladder. A rare entity of
Registration of research studies cystic lesion in the gallbladder, Digest. Dis. Sci. 47 (8) (2002) 1817–1820.
[12] A. Muraoka, N. Watanabe, Y. Ikeda, Y. Kokudo, A. Tatemoto, Y. Sone, et al.,
Ciliated foregut cyst of the gallbladder: report of a case, Surg. Today 33 (9)
None.
(2003) 718–721.
[13] A.S. Bulut, K. Karayalçın, Ciliated foregut cyst of the gallbladder: report of a
Guarantor case and review of literature, Pathol. Res. Int. (2010) 193535.
[14] J.H. Kim, Y.S. Jeon, J.I. Lee, Cilated foregut cyst of the gallbladder: a case report
with US and CT findings, J. Korean Soc. Radiol. 62 (2010) 373–376.
Triki Wissem. [15] A. Giakoustidis, D. Morrison, A. Thillainayagam, Ciliated foregut cyst of the
gallbladder: a diagnostic challenge and management quandary, J.
Gastrointest. Liver Dis. 23 (2) (2014) 207–210.
Acknowledgement
[16] M.J. Lee, J. Salinas, W. Varikatthas, G. Alsnih, A rare gallbladder ciliated foregut
cyst in chronic cholecystitis, Int. J. Surg. Case Rep. 20 (2016) 155–158.
The patient has given informed consent to the publication. [17] P. Agarwal, A. Ahuja, M. Bhardwaj, Ciliated foregut cyst of gallbladder: a first
in childhood and review of literature, Fetal Pediatr. Pathol. 20 (2016) 1–5.
[18] I. Hwang, J. Cho, Ciliated foregut cyst of the gallbladder: a case report and
References literature review, Korean J. Hepatobiliary Pancreat. Surg. 20 (2016) 85–88.
[19] J. Eun Han, M. Hwan Noh, W. Kim, A case of ciliated foregut cyst of the
[1] R.A. Agha, A.J. Fowler, A. Saetta, I. Barai, S. Rajmohan, D.P. Orgill, The SCARE gallbladder, Korean J. Gastroenterol. 67 (2016), 49–45.
statement: consensus-based surgical case report guidelines, Int. J. Surg. 34
(2016) 180–186.
[2] H.E. Robertson, W.J. Ferguson, The diverticula (Luschka’s crypts) of the
gallbladder, Arch. Pathol. (Chic) 40 (1945) 312–333.

Open Access
This article is published Open Access at sciencedirect.com. It is distributed under the IJSCR Supplemental terms and conditions, which
permits unrestricted non commercial use, distribution, and reproduction in any medium, provided the original authors and source are
credited.