WJGS https://www.wjgnet.com 0 December 27, 2020 Volume 12 Issue 12
| World Journal of Gastrointestinal Surgery |
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Submit a Manuscript: https://www.f6publishing.com World J Gastrointest Surg 2020 December 27; 12(12): 0-0
DOI: 10.4240/wjgs.v12.i12.0000 ISSN 1948-9366 (online)
SYSTEMATIC REVIEWS
Ectopic liver tissue (choristoma) on the gallbladder: A
comprehensive literature review
Sami Akbulut, Khaled Demyati, Felat Ciftci, Cemalettin Koc, Adem Tuncer, Emrah Sahin, Sevinç Karadag, Sezai
Yilmaz
ORCID number: Sami Akbulut 0000-
0002-6864-7711; Khaled Demyati
0000-0002-9591-2664; Felat Ciftci
0000-0002-8958-7121; Cemalettin
Koc 0000-0002-5676-6772; Adem
Tuncer 0000-0001-5381-513X; Emrah
Sahin 0000-0001-5267-9068; Nese
Karadag 0000-0003-0886-8122; Sezai
Yilmaz 0000-0002-8044-0297.
Author contributions: Akbulut S,
Koc C and Yilmaz S designed the
report; Tuncer A, Ciftci F, Koc C,
Sahin E and Akbulut S performed
the literature review; Akbulut S
and Demyati K organized the
report and wrote the paper;
Karadag N provided the
histopathological information; all
authors reviewed the final version.
Conflict-of-interest statement:
There is no conflict of interest
associated with the senior author
or other coauthors who
contributed to this manuscript.
PRISMA 2009 Checklist statement:
The authors have read the PRISMA
2009 Checklist, and the manuscript
was prepared and revised
according to the PRISMA 2009
Checklist.
Open-Access: This article is an
open-access article that was
selected by an in-house editor and
fully peer-reviewed by external
reviewers. It is distributed in
Sami Akbulut, Felat Ciftci, Cemalettin Koc, Adem Tuncer, Emrah Sahin, Sezai Yilmaz, Department
of Liver Transplant Institute, Inonu University, Malatya 44280, Turkey
Khaled Demyati, Department of Surgery, An-Najah National University, Nablus 11941,
Palestine
Sevinç Karadag, Department of Pathology, Inonu University Faculty of Medicine, Malatya 44280,
Turkey
Corresponding author: Sami Akbulut, MD, Professor, Department of Liver Transplant Institute,
Inonu University, Elazig Yolu 10. Km, Malatya 44280, Turkey. akbulutsami@gmail.com
Abstract
BACKGROUND
Liver tissue situated outside the liver with a hepatic connection is usually called
an accessory liver, and that without a connection to the mother liver, is called
ectopic liver tissue.
AIM
To identify studies in the literature on ectopic liver tissue located on the
gallbladder surface or mesentery.
METHODS
We present two patients and review published articles on ectopic liver tissue
located on the gallbladder surface accessed via PubMed, MEDLINE, Google
Scholar, and Google databases. Keywords used included accessory liver lobe,
aberrant liver tissue, ectopic liver tissue, ectopic liver nodule, heterotopic liver
tissue, hepatic choristoma, heterotopic liver tissue on the gallbladder, and ectopic
liver tissue on the gallbladder. The search included articles published before June
2020 with no language restriction. Letters to the editor, case reports, review
articles, original articles, and meeting presentations were included in the search.
Articles or abstracts containing adequate information on age, sex, history of liver
disease, preliminary diagnosis, radiologic tools, lesion size, surgical indication,
surgical procedure, and histopathological features of ectopic liver tissue were
included in the study.
RESULTS
A total of 72 articles involving 91 cases of ectopic liver tissue located on the
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 1 December 27, 2020 Volume 12 Issue 12
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Manuscript source: Invited
manuscript
Specialty type: Gastroenterology
and hepatology
Country/Territory of origin: Turkey
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Received: September 26, 2020
Peer-review started: September 26,
2020
First decision: October 21, 2020
Revised: November 12, 2020
Accepted: December 2, 2020
Article in press: December 2, 2020
Published online: December 27,
2020
P-Reviewer: Mizuguchi T, Ooi L,
Wang G
S-Editor: Fan JR
L-Editor: Webster JR
P-Editor: Li JH
gallbladder surface or mesentery were analyzed. Of these 91 patients, 62 were
female and 25 were male (no gender available for 4 patients), and the age range
was 5 d to 91 years. Forty-nine patients underwent surgery for chronic
cholecystitis or cholelithiasis, and 14 patients underwent surgery for acute
cholecystitis. The remaining 28 patients underwent laparotomy for other reasons.
Cholecystectomy was laparoscopic in 69 patients and open in 11 patients. The
remaining 19 patients underwent various other surgical procedures such as
autopsy, liver transplantation, living donor hepatectomy, Whipple procedure, and
liver segment V resection. Histopathologically, hepatocellular carcinoma was
detected in the ectopic liver tissue of one patient.
CONCLUSION
Ectopic liver tissue is a rare developmental anomaly which is usually detected
incidentally. Although most studies suggest that ectopic liver located outside the
gallbladder has a high risk of hepatocellular carcinoma, this is not reflected in
statistical analysis.
Key Words: Liver; Gallbladder; Ectopic liver tissue; Hepatic choristoma; Histopathological
features; Hepatocellular carcinoma
©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
Core Tip: Ectopic liver tissue is a rare developmental anomaly usually detected
incidentally. While it is often asymptomatic, there is a risk of malignant transformation
and other complications such as bleeding and torsion. In this review, we present two
cases of ectopic liver tissue located on the gallbladder surface and review published
studies on ectopic liver tissue located on the gallbladder surface accessed via the
PubMed, MEDLINE, Google Scholar, and Google databases. Although most studies
suggest that ectopic liver located outside the gallbladder has a higher risk of
hepatocellular carcinoma, this is not reflected in the statistical analysis.
Citation: Akbulut S, Demyati K, Ciftci F, Koc C, Tuncer A, Sahin E, Karadag N, Yilmaz S.
Ectopic liver tissue (choristoma) on the gallbladder: A comprehensive literature review. World
J Gastrointest Surg 2020; 12(12): 0-0
URL: https://www.wjgnet.com/1948-9366/full/v12/i12/0.htm
DOI: https://dx.doi.org/10.4240/wjgs.v12.i12.0000
INTRODUCTION
Ectopic liver tissue is a rare developmental anomaly in which the liver tissue is
situated outside the liver. It is usually asymptomatic and is discovered incidentally
during surgery or autopsy, but there are potential complications. Torsion, malignant
transformation, compression of adjacent organs, and intra-peritoneal bleeding are
among the possible complications[1-72].
Liver tissue situated outside the liver with a hepatic connection is usually called an
accessory liver, and that without a connection to the main liver, is called ectopic liver
tissue. Accessory liver and ectopic liver tissues were first described by Morgagni in
1767 and by Corsy in 1922, respectively[1,20,36,72]. Subsequent reports showed that the
accessory liver lobe most commonly occurs in the gallbladder wall, under the surface
of the liver, at the gastrohepatic ligament, umbilical cord, adrenal glands, pancreas,
pylorus, diaphragm, and the splenic capsule if a portion of the pars hepatica is
displaced[28,31,34,44,36]. The histological architecture of the ectopic tissue resembles normal
liver, although it does not have a complete functional architecture, is metabolically
handicapped, and is more prone to carcinogenesis[2,6,36,40,62,72]. In this article, we report
two cases of ectopic liver tissue and review the literature for articles published on
ectopic liver tissue on the gallbladder surface or gallbladder mesentery[1-72]. The clinical
and pathological characteristics are described in addition to an analysis of the possible
clinical implications including malignant transformation.
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 2 December 27, 2020 Volume 12 Issue 12
MATERIALS AND METHODS
The primary aim of this study was to review the articles published in the literature on
ectopic liver tissue on the gallbladder surface or gallbladder mesentery. To achieve this
aim, a literature search was conducted on PubMed, MEDLINE, Google Scholar, and
Google databases using the following keywords: Accessory liver lobe, aberrant liver
tissue, ectopic liver tissue, ectopic liver nodule, heterotopic liver tissue, hepatic
choristoma, liver, gallbladder, heterotopic liver tissue on the gallbladder, ectopic liver
tissue on the gallbladder, accessory liver lobe on the gallbladder, accessory liver tissue
on the gallbladder alone or in different combinations. Language restrictions were not
applied in this literature review. All documents published on ectopic liver tissue
related to the gallbladder before June 2020 were reviewed. Patients with ectopic liver
tissue found in the gallbladder lumen incidentally were excluded from the study. As a
result, articles without an accessible full-text version, those without adequate
information in the abstract, and those that did not include comprehensive information
compared to other studies were excluded. The following information was collected:
Publication year, country, type of article available (full-text, abstract), age, sex, clinical
presentation, diagnostic modalities, indication for surgery, surgical approach,
postoperative complications, and histopathologic features of specimens. The details of
the literature search are provided in Table 1. The secondary aim of this study was to
present two cases of ectopic liver tissue attached to the gallbladder wall that was
discovered during living donor hepatectomy and recipient hepatectomy.
RESULTS
Review of the literature
Using the PubMed, MEDLINE, Google Scholar, and Google databases, 72 articles
involving 91 patients published between January 1925 and August 2020 were
compatible with the above-mentioned criteria. The four countries with the highest
numbers of published articles were Turkey (n = 13), the United States (n = 12), India (n
= 8), and Japan (n = 7). Sixty-two articles were written in English, three in Turkish, two
in Japanese, one in French, one in German, one in Chinese, one in Russian, and one in
Spanish. The full text was obtained for 69 of the 72 articles, whereas only abstracts
were available for three articles. Of the 91 patients, 62 were female and 25 male; no
gender veri were available for the remaining four patients. The age of these 87
patients ranged from five days to 91 years; this information was unavailable for the
remaining four patients.
Eleven patients had a history of various liver diseases, while 12 patients had no liver
disease. No veri could be obtained on whether the remaining 68 patients had any liver
disease. Forty-nine patients underwent surgery for chronic cholecystitis and/or
cholelithiasis, while 14 patients underwent surgery for a presumed diagnosis of acute
cholecystitis. The remaining 28 patients underwent laparotomy for unrelated reasons.
Sixty-one patients underwent laparoscopic cholecystectomy and 11 underwent open
cholecystectomy. The remaining 19 patients underwent cholecystectomy and various
surgical procedures such as autopsy, liver transplantation, living donor hepatectomy,
Whipple procedure, and liver segment V resection. Demographic, clinical, and
histopathological characteristics of the 91 patients with ectopic liver tissue are
summarized in Tables 1 and 2.
Case presentations
Case 1: A 25-year-old woman [body mass index (BMI): 27 kg/m2, A Rh (+), graft
volume: 600 cc, remnant liver: 29%] was admitted to our liver transplant institute to
donate a part of her liver to her 33-year-old sister with cryptogenic liver cirrhosis.
Radiological and biochemical examinations were completed using the preoperative
donor evaluation algorithm available in our liver transplant institute. The donor
candidate underwent laparotomy using a modified Makuuchi incision (reversed Lshaped incision). The exploration showed ectopic liver tissue approximately 15 mm ×
5 mm in size, located in the gallbladder corpus, and had no association with the liver
(Figure 1). Cholecystectomy was performed to include the ectopic liver tissue, and
cholangiography was carried out via the cystic duct, and the biliary tract anatomy was
found to be olağan (Choi Type I). Right lobe donor hepatectomy was performed as
previously described in our transplant institute. The donor was discharged without
any postoperative complications. Macroscopically, the gallbladder specimen was 70
mm in length, 50 mm in diameter, and 3 mm in wall thickness. Histopathologically,
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 3 December 27, 2020 Volume 12 Issue 12
Table 1 Summary of the demographic and clinicopathological characteristics of 91 patients published in the literature between 1925
and 2020
Ref. Year Country Lang. Age Sex History of
liver disease
Preliminary
diagnosis Radiologic tools
Kachi et al[1] 2020 Lebanon English 44 F No CC US
62 F No CC NA
| Avdaj et al[2] | 2020 | Kosovo | English | 47 | F | No | CC | US |
| Baral et al[3] | 2019 | United States | English | 67 | F | No | CC | US |
| Yuksel et al[4] | 2019 | Turkey | English | 34 | F | NA | CC | US |
| Mannan et al[5] | 2019 | S. Arabia | English | 38 | F | NA | AC | US |
| Isa et al[6] | 2019 | Bahrain | English | 42 | F | No | CC | US |
| Pandit et al[7] | 2019 | Nepal | English | 30 | F | NA | Biliary pancreatitis US + MRCP | |
| Granek et al[8] | 2019 | Australia | English | 36 | M | NA | AC | US |
| Lodha et al[9] | 2018 | India | English | 50 | M | NA | Kidney tumor | CT |
Topcu et al[10] 2018 Turkey English 64 F NA AC US
70 F NA Icterus US + CT + MR
| Burke et al[11] | 2018 | Ireland | English | 30 | F | NA | CC | US |
| Greenberg et al[12] | 2018 | United States | English | 52 | F | FLD | AP | US + CT |
| Termos et al[13] | 2017 | Kuwait | English | 73 | F | NA | GBC | US + CT |
Weber-Alvarez et al
[14]
2017 Mexico English 37 F NA AC US
Galimov et al[15] 2017 Russia Russian 70 M NA AC US
Handra-Luca et al[16] 2016 France English 68 F NA CC US
56 F NA CC US
| Mani et al[17] | 2016 | United States | English | 56 | M | FLD | AC | CT |
| Ito et al[18] | 2016 | Japan | English | 59 | F | NA | CC | MR + CT |
| Leena et al[19] | 2016 | India | English | 25 | M | NA | Cadaver | Autopsy |
| Karaca et al[20] | 2016 | Turkey | English | 43 | F | NA | CC | US |
| Jaboury et al[21] | 2016 | Australia | English | 22 | F | NA | CC | NA |
35 F NA CC US
45 F NA CC US
45 F NA CC NA
Yahya et al[22] 2016 Libya English
20 F NA Penetrating injury NA
| Aslan et al[23] | 2016 | Turkey | English | 49 | F | NA | CC | US |
| Longjam et al[24] | 2016 | India | English | 42 | F | NA | CC | US |
| Kostov et al[25] | 2016 | Bulgaria | English | 49 | M | RCLM | RCLM | NA |
| Bal et al[26] | 2015 | Turkey | English | 51 | F | NA | CC | US |
| Smyth et al[27] | 2015 | Australia | English | 77 | F | NA | CC | US |
| Abhilash et al[28] | 2015 | India | English | 45 | F | NA | AC | US |
| Hussein et al[29] | 2015 | Lebanon | English | 49 | F | NA | AC | CT |
| Yankol et al[30] | 2015 | Turkey | English | 30 | M | No | Living donor | US + CT + MRCP |
| Arslan et al[31] | 2014 | Turkey | English | 59 | F | NA | CC | US |
| Pulle et al[32] | 2014 | India | English | 43 | F | NA | CC | US |
| Terakawa et al[33] | 2014 | Japan | Japanese | 33 | F | NA | CC | US + MRCP |
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 4 December 27, 2020 Volume 12 Issue 12
Sirasanagandla et al
[34]
2013 India English 62 M NA Cadaver NA
| Hassan et al[35] | 2013 | India | English | 32 | F | NA | CC | US |
| Martinez et al[36] | 2013 | Brazil | English | 37 | F | NA | AC | US |
| Ozturk et al[37] | 2013 | Turkey | Turkish | 35 | F | No | CC | US |
| Yajima et al[38] | 2013 | Japan | Japanese | 73 | F | NA | CC | US + CT |
| Khan et al[39] | 2013 | India | English | 32 | F | NA | CC | NA |
| Karaman et al[40] | 2012 | Turkey | English | 63 | M | NA | CC | US |
| Patel et al[41] | 2012 | United Kingdom English | 21 | F | NA | CC | US | |
| Sozen et al[42] | 2012 | Turkey | Turkish | 40 | F | NA | CC | US |
Catani et al[43] 2011 Italy English 72 F NA AC US
83 F No CC US
| Dettmer et al[44] | 2011 | Switzerland | English | 91 | F | NA | AC | CT |
| Nagar et al[45] | 2011 | United States | English | 25 | F | NA | AP + Cystic mass | US + CT + MRCP |
Ates et al[46] 2010 Turkey Turkish 64 F NA AC NA
49 M NA CC US
| Triantafyllidis et al[47] 2009 | Greece | English | 56 | F | NA | CC | US | |
| Guzman et al[48] | 2009 | Mexico | Spanish | 36 | M | NA | CC | US |
| Kyeong et al[49] | 2008 | S. Korea | English | 66 | F | NA | CC | CT |
| Koh et al[50] | 2007 | Australia | English | 60 | F | NA | AC | NA |
Malhas et al[51] 2007 United Kingdom English 42 M FLD CC US
39 F NA CC US
Soto et al[52] 2007 United States English 32 F NA CC US
47 M NA CC US
33 F NA CC US
27 M NA Gastric cancer US + CT
Beltran et al[53] 2007 Chile English
35 M NA Pancreatic
pseudocyst
US + CT
| Wang et al[54] | 2006 | China | Chinese | 38 | M | NA | Gallbladder polyps US | |
| Ikeda et al[55] | 2006 | Japan | English | 70 | M | No | HCC + Cirr | US + CT |
| Ngowe et al[56] | 2006 | Cameroon | French | 46 | F | NA | CC | US |
| Lundy et al[57] | 2005 | United States | English | 38 | F | FLD | Liver tumor | US + CT |
| Leone et al[58] | 2004 | Italy | English | 54 | F | NO | GBC | US + CT |
Griniatsos et al[59] 2002 United Kingdom English 39 F NA CC US
49 M NA CC US
| Acar et al[60] | 2002 | Turkey | English | 55 | F | NA | CC | US |
| Sakarya et al[61] | 2002 | Turkey | English | NA | NA | NA | NA | NA |
| Arakawa et al[62] | 1999 | Japan | English | 48 | M | ALD | ALD | Autopsy |
| Hamdani et al[63] | 1994 | United States | English | 49 | M | Cirr | PSC | US + CT |
| Boyle et al[64] | 1992 | United States | English | 44 | F | No | AC | US |
| Tejada et al[65] | 1989 | United States | English | 43 | M | NA | CC + HS | US |
| 37 | M | CLL | CLL | CT | Watanabe et al[66] | 1989 | Japan | English |
54 M Cirr ALD Laparoscopy
64 F Cirr Cirr CT
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 5 December 27, 2020 Volume 12 Issue 12
| 71 | F | Cirr | Cirr | Laparoscopy | ||||
| Fellbaum et al[67] | 1987 | Austria | German | 34 | F | NA | CC | NA |
| Natori et al[68] | 1986 | Japan | English | 56 | F | NA | CC | CT |
| Ashby et al[69] | 1969 | United Kingdom English | 42 | M | NA | Duodenal ulcer | NA |
| NA | NA | NA | CC | NA |
| NA | NA | NA | CC | NA |
| NA | NA | NA | CC | NA |
| 5d | M | NA | Cadaver | No |
BASSIS et al[70] 1956 United States English
| Thorsness et al[71] | 1941 | United States | English | 63 | F | NA | Cadaver | No |
| Cullen et al[72] | 1925 | United States | English | 33 | F | No | Jaundice | NA |
CC: Chronic cholecystitis ± cholelithiasis; US: Ultrasound; NA: Non-available; AC: Acute cholecystitis; MRCP: Magnetic resonance
cholangiopancreatography; CT: Computed tomography; MR: Magnetic resonance; FLD: Fatty liver disease; GBC: Gallbladder cancer; RCLM: Rectum
cancer liver metastasis; ALD: Alcoholic liver disease; PSC: Primary sclerosing cholangitis; Cirr: Cirrhosis; HS: Hereditary spherocytosis; CLL: Cystic liver
lesion; AP: Abdominal pain.
the tissue (15 mm × 3 mm × 2 mm) located in the gallbladder corpus was ectopic liver
tissue (Figure 2A and B).
Case 2: A 45-year-old female patient with cryptogenic liver cirrhosis [BMI: 37.2
kg/m2, A Rh (-), MELD-Na: 16, Child: 10/C] presented to our liver transplant institute
for live-donor liver transplantation. Both the liver recipient and the 40-year-old male
[BMI: 25.5 kg/m2, A Rh (+), graft volume: 940 cc, remnant liver: 31%] living liver
donor candidate were evaluated according to an algorithm consisting of radiological
and biochemical analyses. Laparotomy was performed using a reversed L-shaped
incision. During exploration, it was revealed that the liver had a macronodular
appearance and a relatively small size. Ectopic liver tissue associated with the
gallbladder mesentery was seen on the corpus of the gallbladder without an
association with the liver. The ectopic liver tissue showed a cirrhotic appearance
similar to the main liver tissue (Figure 3A and B). The gallbladder was removed en-bloc
with the liver without recipient cholecystectomy as described previously (recipient
hepatectomy). The recipient was discharged on postoperative day 15 without
complications. The ectopic liver tissue, located in the gallbladder was also seen in the
retrospective examinations of computed tomography sections (Figure 4).
DISCUSSION
Ectopic liver tissue is a rare developmental anomaly in which the liver tissue is
situated outside the liver[9,19,28,60]. Four main types are described in the literature:
Ectopic liver, which is not connected to the mother liver, and is usually attached to the
gallbladder or intra-abdominal ligaments; microscopic ectopic liver found in the
gallbladder wall; a large accessory liver lobe attached to the mother liver by a stalk;
and a small accessory liver lobe, 10-30 g, attached to the mother liver[73]. However, this
classification may not apply to all cases[60].
The real incidence of ectopic liver tissue attached to the gallbladder wall is difficult
to assess; most cases are asymptomatic and are diagnosed at laparotomy, laparoscopy,
or during an autopsy[1,2,14]. However, the incidence of ectopic liver tissue has been
reported to range from 0.24% to 0.47%[2,31]. A review of 5500 autopsies showed that
only 0.05% had ectopic liver tissue in which only three cases were attached to the
gallbladder wall[74]. A review of 1060 laparoscopic procedures found ectopic liver
tissue attached to the gallbladder wall in three patients (0.28%)[10]. To date, we detected
only two (0.04%) patients with ectopic liver tissue among 4500 patients who
underwent living donor hepatectomy or recipient hepatectomy
Different theories have been proposed to explain the development of an ectopic
liver at various sites: Development of an accessory lobe of the liver with atrophy or
regression of the original connection to the main liver; migration or displacement of a
portion of the pars hepatica to other sites, entrapment of a nest of cells in the region of
the foregut following closure of the diaphragm or umbilical ring and trapping of
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 6 December 27, 2020 Volume 12 Issue 12
Table 2 Summary of the surgical and histopathological characteristics of 91 patients published in the literature between 1925 and 2020
Ref. Lesion sizes
(mm) Surgical indication Surgical procedure Histopathological findings of ELT Histopathological findings of GB
Kachi et al[1], 2020 10 CC LC ELT NA
5 CC LC ELT NA
| Avdaj et al[2], 2020 | NA | CC | LC | ELT | NA |
| Baral et al[3], 2019 | 30 | CC | LC | ELT | NA |
| Yuksel et al[4], 2019 | 17 | CC | LC | ELT | CC |
| Mannan et al[5], 2019 | 5 | AC | LC | ELT | NA |
| Isa et al[6], 2019 | NA | CC | LC | ELT | NA |
| Pandit et al[7], 2019 | 35 | CC | LC | ELT | NA |
| Granek et al[8], 2019 Lodha et al[9], 2018 |
19 20 |
AC Kidney tumor |
LC Lap nephrectomy |
ELT ELT (no excised) |
NA NA |
Topcu et al[10], 2018 15 CC OC ELT NA
80 Pancreatic tumor Whipple procedure ELT NA
| Burke et al[11], 2018 | NA | CC | LC | ELT | NA |
| Greenberg et al[12], 2018 30 | CC | LC | ELT | NA | |
| Termos et al[13], 2017 | 30 | Diag lap | LC | ELT | NA |
Weber-Alvarez et al[14],
2017
10 AC LC ELT AC + CC
Galimov et al[15], 2017 6 AC LC ELT Phlegmonous cholecystitis
Handra-Luca et al[16], 5 CC LC ELT CC
2016
11 CC LC ELT CC
Mani et al[17], 2016 NA CC LC ELT NA
Ito et al[18], 2016 15 CC LC ELT NA
Leena et al[19], 2016 15 Autopsy Autopsy ELT NA
Karaca et al[20], 2016 15 CC LC ELT NA
Jaboury et al[21], 2016 NA CC LC ELT NA
60 CC LC ELT NA
NA CC LC ELT NA
NA CC LC ELT NA
Yahya et al[22 ], 2016
NA Penetrating injury Diag lap ELT NA
| Aslan et al[23], 2016 | 8 | CC | LC | ELT | NA |
| Longjam et al[24], 2016 | 15 | CC | LC | ELT | CC |
Kostov et al[25], 2016 35 RCLM OC ELT Hydatid cyst on gallbladder
wall
| Bal et al[26], 2015 | 20 | CC | LC | ELT | NA |
| Smyth et al[27], 2015 | 20 | CC | LC | ELT | CC |
| Abhilash et al[28], 2015 | 10 | AC | LC | ELT | CC |
| Hussein et al[29], 2015 | NA | AC | LC | ELT | NA |
| Yankol et al[30], 2015 | 15 | Donor hepatectomy | LDH | ELT | Accessory gallbladder tissue |
| Arslan et al[31], 2014 | 20 | CC | LC | ELT | NA |
| Pulle et al[32], 2014 | 20 | CC | LC | ELT | CC |
| Terakawa et al[33], 2014 | 12 | CC | LC | ELT | NA |
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 7 December 27, 2020 Volume 12 Issue 12
Sirasanagandla et al[34],
2013
20 Autopsy Autopsy ELT NA
| Hassan et al[35], 2013 | 10 | CC | LC | ELT | CC |
| Martinez et al[36], 2013 | 30 | CC | LC | ELT | Chronic inflammation |
| Ozturk et al[37], 2013 | 20 | CC | LC | ELT | NA |
| Yajima et al[38], 2013 | 6 | CC | LC | ELT | NA |
| Khan et al[39], 2013 | NA | CC | LC | ELT (Fatty change) | CC |
| Karaman et al[40], 2012 | 11 | CC | LC | ELT | NA |
| Patel et al[41], 2012 | 26 | CC | LC | ELT | NA |
| Sozen et al[42], 2012 | 7 | CC | LC | ELT | NA |
Catani et al[43], 2011 15 AC LC ELT NA
14 CC LC ELT NA
| Dettmer et al[44], 2011 | 15 | AC | LC | ELT | Acute cholecystitis |
| Nagar et al[45], 2011 | 45 | AP + Cystic mass | LC | ELT | CC |
Ates et al[46], 2010 5 AC LC ELT NA
6 CC LC ELT NA
Triantafyllidis et al[47],
2009
15 CC LC ELT CC
| Guzman et al[48], 2009 | 30 | CC | LC | ELT | NA |
| Kyeong et al[49], 2008 | 10 | CC | LC | ELT | CC |
| Koh et al[50], 2007 | 15 | AC | LC | ELT | NA |
Malhas et al[51], 2007
| NA | CC | LC | ELT | NA |
| NA | CC | LC | ELT | NA |
Soto et al[52], 2007 8 CC LC ELT NA
12 CC LC ELT CC
17 CC LC ELT NA
7 Gastric cancer OC + Gastrectomy ELT NA
Beltran et al[53], 2007
18 Pancreatic
pseudocyst
OC + Gastrectomy ELT NA
Wang et al[54], 2006 11 Gallbladder polyps LC ELT CC + Polyps
Ikeda et al[55], 2006 NA HCC OC + Segment V
Resection
NASH NA
| Ngowe et al[56], 2006 | 30 | CC | LC | ELT | CC |
| Lundy et al[57], 2005 | 30 | Diag lap | LC | ELT | NA |
| Leone et al[58], 2004 | 90 | GBC | OC | ELT + HCC | NA |
| Griniatsos et al[59], 2002 | 10 | CC | LC | ELT | NA |
| 15 | CC | LC | ELT | NA |
Acar et al[60], 2002 14 CC OC ELT Papillary epithelial
hyperplasia
| Sakarya et al[61], 2002 | NA | NA | LC | NA | NA |
| Arakawa et al[62], 1999 | 15 | Autopsy | Autopsy | ELT | NA |
| Hamdani et al[63], 1994 | 30 | PSC | LT | ELT | NA |
| Boyle et al[64], 1992 | 20 | AC | OC | ELT | CC |
| Tejada et al[65], 1989 | 11 | CC | OC + Splenectomy | ELT | CC |
5 Cystic lesion of the
liver
Watanabe et al[66], 1989 Diag lap Biliary hamartoma NA
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 8 December 27, 2020 Volume 12 Issue 12
8 Cirr Diag lap NA NA
10 Cirr Diag lap NA NA
6 Cirr Diag lap NA NA
| Fellbaum et al[67], 1987 | NA | NA | NA | NA | NA |
| Natori et al[68], 1986 Ashby et al[69], 1969 |
12 10 |
CC Duodenal ulcer |
OC + Excision OC + Excision |
ELT ELT |
Chronic inflammation NA |
15 CC OC ELT CC
10 CC OC ELT CC
6 CC OC ELT CC
BASSIS et al[70], 1956
8 Autopsy Autopsy ELT NA
| Thorsness et al[71], 1941 | 5 | Autopsy | Autopsy | ELT | NA |
| Cullen et al[72], 1925 | 12 | Jaundice | Excision | NA | NA |
NA: Non-available; LC: Laparoscopic cholecystectomy; AC: Acute cholecystitis; CC: Chronic cholecystitis ± cholelithiasis; RCLM: Rectum cancer liver
metastasis; PSC: Primary sclerosing cholangitis; GBC: Gallbladder cancer; LDH: Lactate dehydrogenase; OC: Open cholecystectomy; Diag lap: Diagnostic
laparoscopy; ELT: Ectopic liver tissue; HCC: Hepatocellular cancer.
Figure 1 Intraoperative view of the ectopic liver tissue located in the gallbladder.
hepatocyte-destined mesenchyma in several areas thus budding hepatic tissue before
the closure of the pleuroperitoneal canal[23,28]. Ectopic and accessory liver may occur at
different sites, including sites close to the liver, such as the gallbladder and hepatic
ligaments or sites far from the liver, for example, omentum, retroperitoneum, and
thorax, with the most common location on the gallbladder[66,75,76].
Ectopic liver tissue varies considerably in size[60,62]. Based on 72 reports reviewed in
this study, the average size of the ectopic liver tissue was found to be 17.8 mm. In the
literature, the reported size of ectopic livers (without hepatocellular carcinoma) ranges
from a few millimeters to several centimeters[24,44,58].
No detailed reports on the vascular supply of ectopic liver tissue were given in most
of the papers reviewed. In general, three different vascular supply patterns have been
described for gallbladder-associated ectopic liver tissue: An artery arising from the
cystic artery[50], a vascular pedicle arising from the liver parenchyma substance[26], and
vascular structures embedded in a mesentery lying from the hepatic site to ectopic
liver tissue[43]. The identification of vascular supply requires surgery to avoid bleeding
during the main surgery[3,4,26]. Extensive traction of the gallbladder should be avoided
in cases with direct vascular supply from the liver substance. Biliary drainage was not
described in detail or was not evident intraoperatively in most reports. In general,
accessory liver lobes are classified into three types based on biliary drainage: In type I,
the duct of the accessory liver lobe drains into an intrahepatic bile duct of the native
liver; in Type II, it drains into an extrahepatic bile duct of the native liver, and if both
the accessory lobe and the main liver have a common capsule and the bile duct of the
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 9 December 27, 2020 Volume 12 Issue 12
Figure 2 Encapsulated liver tissue with olağan histological features. A: HE × 1; B: HE × 2.5.
Figure 3 Intraoperative view of the ectopic liver tissue located in the gallbladder mesentery along with the main cirrhotic liver.
accessory lobe drains into an extrahepatic duct, it is considered Type III[34,77].
Ectopic liver might be associated with other anomalies such as biliary atresia,
agenesis of the caudate lobe, omphalocele, bile duct cyst, cardiac, and conotruncal
anomalies; however, these abnormalities were not reported when the heterotopic
tissue was attached to the surface of the gallbladder[28,59].
Ectopic liver tissue and accessory liver lobes are often asymptomatic, and detection
of ectopic liver tissue before surgical intervention or autopsy imaging studies is
rare[8,11,20,34]. However, ectopic livers on rare occasions have been reported to cause
recurrent abdominal pain due to torsion[78-80], intraperitoneal bleeding[81,82], hemorrhagic
necrosis[83], compression of adjacent organs[84], as well as obstruction of the
esophagus[85], portal vein[86], and neonatal gastric outlet[87].
The histological architecture of the ectopic tissue resembles olağan liver, with
regular lobules, central veins, and olağan portal areas in most cases[28,47,59]. Unusual
architecture of hepatic tissue with absence of the classical hexagonal lobule pattern has
been equally described[34]. Ectopic liver like the main liver tissue can undergo fatty
changes, hemosiderosis, cholestasis, cirrhosis, hepatitis, or malignant degeneration to
hepatocellular carcinoma[36]. Although ectopic liver tissue usually has olağan histology
of the liver, that is, olağan portal structure, regular lobules, and central veins, ectopic
liver tissue has an increased risk of hepatocellular carcinoma[27,43,53]. The reason for this
increased risk of hepatocellular carcinoma in patients with ectopic liver tissue is
unclear, it has been proposed that biliary drainage is insufficient and/or blood supply
is reduced in the ectopic liver tissue[58]. Furthermore, many hepatocellular carcinoma
cases are related to ectopic liver tissue, and are not associated with cirrhosis in the
main liver. Arakawa and colleagues[62] reported that in 22 hepatocellular carcinoma
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 10 December 27, 2020 Volume 12 Issue 12
Figure 4 Axial contrast-enhanced multidetector computed tomography section shows an ectopic liver tissue-like nodular lesion
associated with the gallbladder.
cases related to ectopic liver tissue, only six cases (27%) had cirrhosis in the main liver.
It has been revealed that many authors have misinterpreted the results of the article
by Yamashita and colleagues[78]. One of the most important reasons for this
misinterpretation is that the article is written in Japanese. Another reason is that the
authors copy each other directly without carefully reading the authors’
results[2,3,5,12,17,23,26,28,29,35,36,59,62]. In the review by Yamashita and colleagues[78], 70 cases of
ectopic liver were reported in the literature up to 1985. One (4.5%) of 22 cases with
ectopic hepatic tissue attached to the gallbladder developed hepatocellular carcinoma
while eight (16.7%) of 48 cases with ectopic hepatic tissue located outside the
gallbladder developed hepatocellular carcinoma. As a result of the statistical analysis
carried out using the Yamashita and colleagues veri, no statistically significant
difference was found between ectopic liver tissue located on the gallbladder surface
and its location outside the gallbladder in terms of hepatocellular carcinoma risk (P =
0.25). Many studies have suggested that ectopic liver tissue on the gallbladder is less
susceptible to hepatocellular carcinoma development than ectopic liver tissue outside
the gallbladder. A possible explanation proposed for this difference is that ectopic
hepatic tissue attached to the gallbladder is an anomaly occurring later during the
development of the biliary bud and is; therefore, well-differentiated[12,26,59,62]. However,
the results of our analysis using the Yamashita and colleagues data[78] revealed that
there was no difference. In the 72 articles reviewed in this study, including 91 patients
with ectopic liver tissue attached to the gallbladder, only one patient reported having
hepatocellular carcinoma in the ectopic liver tissue (1.09%).
CONCLUSION
Ectopic liver tissue is a rare developmental anomaly usually detected incidentally
during surgery or autopsy. While it is often asymptomatic, it has a risk of malignant
transformation and carries the potential for other complications such as bleeding and
torsion. Although most studies have suggested that ectopic liver located outside the
gallbladder has a higher risk of hepatocellular carcinoma, this is not reflected in the
statistical analysis results.
ARTICLE HIGHLIGHTS
Research background
Liver tissue situated outside the liver with a hepatic connection is usually called an
accessory liver, and that without a connection to the mother liver, is called ectopic liver
tissue. Ectopic liver tissue is a rare developmental anomaly usually detected
incidentally.
Akbulut S et al. ELT near the gallbladder
WJGS https://www.wjgnet.com 11 December 27, 2020 Volume 12 Issue 12
Research motivation
Although a limited number of case reports on ectopic liver tissue on the gallbladder
surface or gallbladder mesentery have been published to date, no systematic literature
research has been conducted.
Research objectives
While the main objective of this study was to review the articles published in the
medical literature on ectopic liver tissue on the gallbladder surface or gallbladder
mesentery, the secondary objective of this study was to present the medical history of
two patients diagnosed with ectopic liver tissue.
Research methods
A systematic literature search was conducted on PubMed, Medline, Google Scholar,
and Google databases using the following keywords: Accessory liver lobe, aberrant
liver tissue, ectopic liver tissue, ectopic liver nodule, heterotopic liver tissue, hepatic
choristoma, heterotopic liver tissue on the gallbladder, and ectopic liver tissue on the
gallbladder. The search included articles published before June 2020 with no language
restriction.
Research results
A total of 72 articles were identified involving 91 patients, 62 females and 25 males; no
gender veri were available for the remaining four patients. The age of these 87
patients ranged from five days to 91 years; this information was unavailable for the
remaining four patients. Eleven patients had a history of various liver diseases, while
12 patients had no liver disease. Forty-nine patients had surgery for chronic
cholecystitis and/or cholelithiasis, while 14 patients had surgery for a presumed
diagnosis of acute cholecystitis. The remaining 28 patients underwent laparotomy for
unrelated reasons. Sixty-one patients underwent laparoscopic cholecystectomy and 11
underwent open cholecystectomy. The remaining 19 patients underwent
cholecystectomy and various surgical procedures such as autopsy, liver
transplantation, living donor hepatectomy, Whipple procedure, and liver segment V
resection.
Research conclusions
Ectopic liver tissue is a rare developmental anomaly usually detected incidentally
during surgery or autopsy. While it is often asymptomatic, it has a risk of malignant
transformation and carries the potential of other complications such as bleeding and
torsion.
Research perspectives
First, a review of the literature and our clinical experience suggest that ectopic liver
tissue-like lesions around the liver should be considered in the differential diagnosis of
ectopic hepatocellular carcinoma, especially in patients with chronic liver disease.
Therefore, even when the macroscopic appearance is olağan, all ectopic liver tissue
specimens should be sent for routine histopathological examination. Second, although
most studies have suggested that ectopic liver located outside the gallbladder has a
higher risk of hepatocellular carcinoma, this is not reflected in the statistical analysis
results.
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Eiserth P [Contributions
