Case ReportEndometrioma of the Liver: A Case Report and Review ofthe Literature

Prachi Rana ,1 Shida Haghighat,1 and Hyosun Han2

1Department of Internal Medicine, Los Angeles County Hospital/University of Southern California, USA2Department of Gastroenterology and Liver Diseases, Los Angeles County Hospital/University of Southern California, USA

Correspondence should be addressed to Prachi Rana; prachi.rana@med.usc.edu

Received 11 March 2019; Accepted 14 May 2019; Published 4 June 2019

Academic Editor: Melanie Deutsch

Copyright © 2019 Prachi Rana et al. This is an open access article distributed under the Creative Commons Attribution License,which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Hepatic endometriosis is a rare form of endometriosis first described by Finkel in 1986. A thorough review of the literature revealed28 cases of hepatic endometriosis. This unusual condition offers several diagnostic challenges due to its variable appearance onimaging and need for histologic analysis to establish a definitive diagnosis. We present a 42-year-old female initially treated forpresumed hydatid cyst that was later found to be endometriosis in the liver. The case highlights the importance of consideringendometriosis in the differential for a patient presenting with a solitary liver mass regardless of age and previous history ofendometriosis.

1. Introduction

Endometriosis is characterized by the presence of endome-trial tissue outside of the uterine cavity. It is a benign con-dition most commonly noted in the uterus, fallopian tubes,ovaries, and local pelvic peritoneum. Atypical endometriosis,when the condition is found in extrapelvic regions, is rare[1]. While uncommon, atypical endometriosis has beendescribed in remote sites including the GI tract, diaphragm,skin, lung, pleura, kidney, and pancreas.The only organ in theabdomen that is refractory to endometriosis is the spleen [2].

Hepatic endometriosis is one of the most rare forms ofextrapelvic endometriosis, first described in 1986 [3]. Only 28cases have been reported in the English literature. We hereinpresent the 29th case of hepatic endometriosis, a 42-year-old female initially treated for presumed hydatid cyst thatwas later found to be endometriosis in the liver. This rarecondition offers several diagnostic challenges. We offer anexhaustive review of the literature focusing on advances in theclinical manifestation, patient characteristics, pathogenesis,and diagnostic workup of the condition.

2. Case Presentation

A 42-year-old multiparous woman presented with episodic,severe right upper quadrant pain associated with nausea and

vomiting. Her past surgical history included a hysterectomyand left oophorectomy for unclear reasons. Several monthsprior she presented to another hospital for similar symptomsandwas diagnosedwith a hepaticmass. Physical examinationdemonstrated right upper quadrant tenderness without anypalpable masses. Liver function and viral serologies forhepatitis B andCwere normal. Tumormarkers demonstratednormal CA 19-9 and AFP, with mildly elevated CA-125 40U/mL (normal <38U/mL).

Computed tomography with intravenous contrastshowed a 3.2cm x 4cm x 1.8cm multiseptated cystic lesionin the left hepatic lobe and an ill-defined heterogeneoushyperdensity within the peripheral right hepatic lobemeasuring 3cm x 1.3cm (Figure 1). Ultrasound-guided fineneedle aspiration and core biopsy of the left hepatic lesionwere inconclusive.

Further workup revealed a positive Echinococcal IgGantibody and she was started on Albendazole for a presumedhydatid cyst. After completion of therapy, she was sched-uled for complete left lateral hepatic resection. However,she presented again several weeks later with progressiveright upper quadrant pain. At this time repeat computedtomography redemonstrated the left hepatic mass which wasunchanged in size but did not show the right hepatic lesion.Imaging also revealed a new pericardial effusion that was

HindawiCase Reports in HepatologyVolume 2019, Article ID 4734606, 8 pageshttps://doi.org/10.1155/2019/4734606

2 Case Reports in Hepatology

Figure 1: Computed tomography showing 3.2 x 4.0 x 1.8 cmmultiseptated cystic lesion in the left hepatic lobe.

Figure 2: Computed tomography showing small pericardial effu-sion.

not present on previous imaging (Figure 2). Her liver testswere the following: AST 485 U/L (normal 10-40 U/L), ALT308 U/L (normal 5-40 U/L), ALP 50 U/L (normal 35-104U/L), and total bilirubin 0.5 mg/dL (normal <1.0 mg/dL).Given the concern for pericardial involvement, she urgentlyunderwent a laparoscopic left partial hepatectomy (segmentII and partial segment III). The postoperative course wasuneventful. Final pathology was consistent with hepaticendometriosis (Figures 3 and 4). After 2 months of follow-up, the patient was asymptomatic and liver tests normalized.She was started onmedroxyprogesterone acetate and remainswell to date.

3. Discussion

Endometriosis is a common gynecologic disease charac-terized by the presence of endometrial glands and stromaoutside of the uterus. It affects 5-15% of women of reproduc-tive age. Pelvic endometriosis involves the ovaries, fallopiantubes, uterine ligaments, Pouch of Douglas, and surround-ing peritoneum [4]. A more rare form of endometriosis,extrapelvic, includes involvement of gastrointestinal tract,urinary system, thoracic cavity, kidneys, and pancreas. Theexact prevalence of extrapelvic endometriosis is unknownbutis thought to present in an older populationwith amedian ageof 34-40 years [4].

Figure 3: Low-power view of the interface between an endometri-otic nodule and liver parenchyma. Notice the large endometrialtype glands surrounded by endometrial type stroma in the superiorportion of the image.These are separated from the liver parenchyma(lower right portion) by a band of fibrosis. The liver parenchymadisplays macrovesicular steatosis.Hematoxylin & Eosin, 40x.

Figure 4: High-power view of an endometriotic nodule.Endometrioid type glands of varying shape and size showcharacteristic columnar lining, within a cellular endometrial typestroma. The inset shows a portal tract for comparison (from top tobottom: vein, artery, bile ducts); notice the small round shape of thebile ducts and lack of cellular stroma. Hematoxylin & Eosin, 200x.

There is no clear consensus or unifying theory of theexact pathophysiology of extrapelvic endometriosis. Severaltheories have been proposed; however no theory aloneaccounts for the development of extrapelvic endometriosis,suggesting a multifactorial nature to the disease.

The classic theory of retrograde menstruation proposesthat reflux of endometrial fragments through the fallopiantubes duringmenstruation results in implantation of the peri-toneal cavity. Though retrograde menstruation is a commonphenomenon seen in up to 90% of healthy women, not all ofthese women develop extrapelvic endometriosis [5].

The coelomic metaplasia theory suggests thatendometriosis develops from metaplasia of the peritonealepithelium possibly due to environmental or geneticfactors [5, 6]. The induction theory suggests that defectsin embryogenesis give rise to endometrial like tissue. TheMullerian ducts give rise to the female genitourinary tract.

Case Reports in Hepatology 3

In males, this structure dissolves under the influence of anti-Mullerian hormone. However remnants of this structuremay persist and differentiate later in life into endometrioticlike tissue due to the presence of excesses in endogenousor exogenous estrogen as is seen in men with chronic liverdisease and prostate cancer [5, 7].

While these theories account for endometriosis withinthe peritoneal and pelvic cavity and provide some insightinto its pathogenesis in men, they do not account for thecases of disseminated endometriosis seen in cases of lymphnodes, thoracic cavity, and liver involvement, as seen in ourpatient. Whether these original cells originate in the uterusor the peritoneal cavity, the theory that endometriotic tissuedisseminates through lymphatic spread offers a plausibleexplanation for the manifestation of hepatic endometriosis[5].

A thorough review of the literature revealed 28 casesof hepatic endometriosis. Our report adds one case to thisrare clinical finding; herein we present the twenty-ninthcase of hepatic endometriosis. Tables 1 and 2 summarize thepreviously reported cases and ours, comparing the presen-tation, imaging, treatment, and pathologic features. In thisreview, the patient age ranged from 21 to 62 years, with amean of 41.5 years. Of the 19 cases that reported parity, tenwere nulligravid and nine were either uni- or multiparous,thus demonstrating that pregnancy and childbirth have nobearing on hepatic endometriosis. Six of 29 (21%) patientswere postmenopausal, thus showing this condition is notlimited to women of reproductive age and that the diagnosisshould be considered in postmenopausal women. Twelveof 29 (41%) had a prior history of endometriosis; thus thediagnosis should not be limited only to patients with a knownhistory of endometriosis. A significant portion of thesepatients had prior abdominopelvic surgery—at least half(51%) had prior pelvic surgery, and 41% had a hysterectomy,suggesting that endometrial tissue seeding during surgerylater resulted in the development of hepatic endometriosis.Themajority (90%) of patients described in the literature hadepigastric or right upper quadrant pain; only two patientscomplained of characteristic cyclic pain related to menses.Only three patients were asymptomatic and their conditionwas diagnosed incidentally. In one peculiar case, however, thepatient presented with flu-like symptoms and right shoulderpain, misdiagnosed as pneumonia initially [8].

Abdominal US, CT, and MRI are the imaging modalitiesmost frequently used. Typical US findings include well-defined cystic masses with solid components and septations.The majority of CT reports show low density, heterogenouscystic lesions that are either nonenhancing or poorly enhanc-ing. Calcifications have been reported along with irregularsoft tissue components but can be variable. Finally, MRIusually demonstrates signal intensity on T1- and T2-weightedimages, similar to that of normal endometrium. However,because endometrial implants can exhibit various degreesof hemorrhage due to hormonal stimulation, implants maydemonstrate a spectrumof appearances depending on the ageof the hemorrhage but can be variable [9].

Of the 19 cases that reported lab values, 79% had normalliver tests. Three cases exhibited mild transaminitis, and afourth case, ours, had markedly elevated transaminases withAST 485 U/L and ALT 305 U/L.

Excluding two, all patients underwent surgery for treat-ment. The most common surgery was hepatectomy vialaparotomy (59%). Other surgical techniques included ultra-sonic cyst manipulation. In the two nonsurgical cases,those patients were treated with danazol alone [9, 10].Tumor size ranged from 1 to 30cm, with mean tumor size9.8 cm.

The final diagnosis can only be made by histopathologicanalysis. The differential diagnosis includes both benignand malignant conditions, as echinococcal cyst, abscess,hematoma, cystadenoma, and malignant cystic neoplasm,such as cystadenocarcinoma or metastatic disease. Methodof diagnosis was largely by histologic analysis after surgeryin 90% of cases. Only four patients underwent CT-guidedpercutaneous biopsy prior to surgery, with only three yieldinga diagnosis of endometriosis and one case, ours, yieldinginconclusive results. Histopathologic examination of thetumors was consistent with endometriosis as evidencedlargely by fibrous capsules with internal epithelial liningcontaining endometrial glands and stroma. Furthermore,although malignant transformation of endometriosis is arare event, occurring commonly in the ovary, there weretwo cases of malignancy reported, one adenosarcoma andone low-grade endometrial stroma sarcoma [11, 12]. Ofthe eight cases that reported on immunostaining, all eightcases were positive for estrogen and progesterone recep-tor, consistent with endometriosis. Five cases reported onfurther immunohistochemistry markers that included CD10(in endometrial tissue) and/or CK7 (in glandular tissue)[13–17].

Hepatic endometriosis is a rare form of endometriosis.This unusual condition offers several diagnostic challengesbut should be considered in the differential in any femalepresenting with a solitary hepatic mass, regardless of age andprevious history of endometriosis.

Consent

Verbal informed consent was obtained from the patient(s) fortheir anonymized information to be published in this article.

Disclosure

Institutional Mailing Address: LAC + USC Medical Center,1200 N. State St., CT A7D - GME, Los Angeles, CA 90033.

Conflicts of Interest

The authors declared no potential conflicts of interest withrespect to the research, authorship, and/or publication of thisarticle.

4 Case Reports in HepatologyTa

ble1:Patie

ntcharacteris

tics,presentatio

n,andtre

atmento

fcaser

eportsof

hepatic

endo

metrio

sis.

Author,Year

Age

Parity

Pre/Po

st-Menop

ausal

PriorP

elvic

Surgery

Hysterectom

yHistoryof

Endo

metrio

sisSymptom

sMetho

dof

Diagn

osis

Treatm

ent

Asran,

2010

61Unk

nown

Post

Salpingo-

ooph

orectomy

Yes

Yes

Postprandialepigastric

pain

CTgu

ided

percutaneous

liver

biop

syN/A

Bouras,2013

35Nullip

arou

sPre

No

No

No

Recurrent,interm

ittent

epigastricpain

Surgery

Llateralh

epaticsectionectom

yby

laparotomy

Chun

g,1998

40Multip

arou

sPre

Lovarian

cyste

ctom

yNo

Yes

Asym

ptom

atic

Surgery

Cystenucleation

DeR

iggi,2016

27Nullip

arou

sPre

No

No

No

Painlessabdo

minalmass

Surgery

Lhepatectom

yby

laparotomy

Fink

el,1986

21Uniparous

Pre

Lfallo

pian

tube

cyst

removal

No

No

Episo

dics

harp,epigastric

pain

associated

with

nausea

andvomiting

notrelated

tomenses

Surgery

Cystenucleation+Danazol

Fluegen,

2013

32Nullip

arou

sPre

No

No

No

RUQpain

Surgery

Ultrason

icperic

ystectom

y

Goldsmith

,200

948

Nullip

arou

sN/A

Salpingo-

ooph

orectomy

Yes

Yes

Relap

sing/remitting

chronicR

UQpain

Surgery

Non

anatom

icresection,

laparotom

y,ultrason

iccystaspiratio

nGroves,2003

52N/A

N/A

Oop

horectom

yYes

No

RUQpain

Surgery

Rhepatectom

y

Hertel,2012

44N/A

N/A

Oop

horectom

yPartial

noSudd

enon

setu

pper

abdo

minalpain

Surgery

Partialh

epatectomy

Huang

,2002

56N/A

Post

Salpingo-

ooph

orectomy

Yes

Yes

Interm

ittentepigastric

pain

notassociatedwith

menses

Surgery

Lhepatic

lobectom

yby

laparotomy

Inal,200

025

N/A

Pre

No

No

Yes

Pelvicpain,m

assa

ndrectal

hemorrhage

Percutaneous

CTgu

ided

biop

syDanazol

Jelovsek,200

452

Uniparous

Post

Salpingo-

ooph

orectomy

Yes

Yes

Flulik

esym

ptom

s,pleuritic

chestp

ain

Surgery

Leup

rolid

e,then

resectionvialaparotom

y

Keramidaris,

2018

40Multip

arou

sPre

No

No

No

Asym

ptom

atic,incidental

Surgery

Cyste

ctom

yby

laparotomy

Khan,

2002

31N/A

N/A

Hysterectom

y,salpingo-

ooph

orectomy

Yes

Yes

Malaise,abd

ominal

diste

ntion

Surgery

Rhepatectom

y+goserelin

Khan,

2002

61N/A

Post

No

No

Yes

RUQpain

Surgery

Rhepatectom

y

Liu,2015

36Uniparous

Pre

No

No

No

RUQpain

priorto

menstr

uatio

nSurgery

Peric

ystectom

y

N'Se

nda,2002

54Uniparous

Post

Hysterectom

y,oo

phorectomy

Yes

No

RUQpain

for1

year

Surgery

Righth

epatectomyby

thoracolaparotom

y

Nezhat,2005

(1)

36Nullip

arou

sN/A

No

No

No

Cyclice

pigastric

pain

for1

year

Surgery

Cystremovalby

CO2laserlaparoscopically

Nezhat,2005

(2)

30Nullip

arou

sN/A

No

No

Yes

Chronicp

elvicpain,

dysm

enorrhea,and

painful

bowelmovem

ents

Surgery

Laparoscop

icremovalof

liver

mass

Reid,2003

46Nullip

arou

sN/A

Oop

horectom

yYes

yes

RUQpain

Surgery

Rhepatectom

y+goserelin

Rivkine,2013

51Multip

arou

sN/A

Hysterectom

yYes

noEp

igastricpain,vom

iting

Surgery

Llobectom

yby

laparotomy

Roesch-D

ietlan,

2011

25Nullip

arou

sPre

No

No

No

Relap

sing/remittingRU

Qpain

Surgery

Incidentallyfoun

ddu

ringlaparoscop

iccholecystectom

y,tre

ated

with

danazol

Rovati,

1990

37Nullip

arou

sPre

No

No

Yes

Chronic,acyclic

epigastric

pain

Surgery

Segm

entectom

yby

laparotom

y+Danazol

Case Reports in Hepatology 5

Table1:Con

tinued.

Author,Year

Age

Parity

Pre/Po

st-Menop

ausal

PriorP

elvic

Surgery

Hysterectom

yHistoryof

Endo

metrio

sisSymptom

sMetho

dof

Diagn

osis

Treatm

ent

Schu

ld,2011

39Uniparous

Pre

No

No

No

RUQpain,cou

ghSurgery

Segm

entectom

y,transdiaph

ragm

atic

pulm

onarywedge

resection

Sherif,

2016

44N/A

Pre

Hysterectom

yYes

Yes

RUQpain

andvomiting

CTgu

ided

core

biop

sySegm

entectom

y

Tuech,2003

42Nullip

arou

sN/A

No

No

No

Chronic,acyclic

epigastric

pain

Surgery

Deroo

fing&cyste

ctom

y

Verbeke,1996

(1)

34N/A

Pre

No

No

No

Acutea

bdom

enSurgery

Rhemihepatectomy

Verbeke,1996

(2)

62N/A

Post

Yes

No

No

RUQpain

Surgery

Cholecystectom

y,Lhepatectom

y

Rana,2019

42Multip

arou

sPre

Hysterectom

y,L

ooph

orectomy

Yes

No

Severe

RUQpain,N

/VSurgery

Lpartialh

epatectomy

6 Case Reports in HepatologyTa

ble2:Im

agingfeatures

ofcase

repo

rtso

fhepaticendo

metrio

sis.

Author,Year

US

CTMRI

Asran,

2010

N/A

Multip

le,irr

egularlyshaped,

heterogeneou

s,lowdensity

lesio

nsscatteredthroug

hout

theliver

N/A

Bouras,2013

N/A

10cm

cysticlesio

nwith

afattycompo

nent

andcalcificatio

ns10cm

cysticlesio

nwith

afattycompo

nent

and

calcificatio

ns

Chun

g,1998

6.4cm

x3cm

x2.5cm

septated

cyst

Lowdensity

hepatic

cyst,

with

undu

latin

gwallbut

noobviou

sseptatio

nsN/A

DeR

iggi,2016

N/A

30cm

hepatic

cytsin

theL

love

reaching

segm

entsIV,V

,VIII

Fink

el,1986

12.5x12x9.5

cmcysticmassinLlobe

with

possibleseptations

12cm

smoo

th-w

alledcysticlesio

nwith

out

septations

N/A

Fluegen,

2013

N/A

N/A

9.5cm

x12cm

lobu

lated

cystin

segm

entsIV,V

,VIII

Goldsmith

,200

99x11cm

cysticm

assinsegm

entIV.

The

wallapp

earedthickwith

complex

septae.

N/A

11x13cm

cysticm

assinsegm

entsIV

andVIIIw

ithincompletes

eptatio

ns

Groves,2003

Bilaterallesio

ns,largestin

Rpo

sterio

rlobe

12x9cm

N/A

N/A

Hertel,2012

N/A

N/A

9.5x9.1x

11.2cm

cysticmassw

ithathickened

wallinR

hepatic

lobe

Huang

,2002

N/A

9x6cm

wellcirc

umscrib

edcysticmass

with

irregular

softtissuec

ompo

nents

N/A

Inal,200

0

Roun

d,well

defin

edandheterogeneou

sinclu

ding

anecho

iccysticandecho

genic

solid

compo

nentsw

ithseptations

and

solid

compo

nents

Roun

d,wellcirc

umscrib

edheterogeneou

smassw

ithseptations.Fine

punctate/nod

ular

calcificatio

nsatthe

perip

hery

ofthelesion

Alobu

latedbu

twell-d

emarcatedsubcapsularm

assin

thep

osterio

rsegmento

fRlobe

oftheliver

Jelovsek,200

4N/A

11x7cm

mass

N/A

Keramidaris,

2018

Largec

ystic

lesio

nbetweenLandRlobe

oftheliver

none

Multiseptatedcysticlesio

n10.3x7.8x7.7cm

intheL

lobe,

segm

entsIV,II,III

Khan,

2002

Largem

assinRlobe

andsm

allinLlobe

largen

on-enh

ancing

lobu

lated

massinR

lobe

andmassinLlobe;portalvein

thrombo

sisN/A

Khan,

2002

N/A

Largem

asso

ccup

ying

thee

ntire

Rlobe

N/A

Liu,2015

6cm

lesio

nin

Llobe

(segmentIII)

6.5x6cm

loculatedcysticlesio

nin

segm

entIII,w

allw

iththickcomplex

septae

N/A

N'Se

nda,2002

N/A

Hugeh

eterogeneous

hypo

densem

ass

partially

enhanced

after

contrast

injection;

cysticchangesw

/fluidlevels

Heterogeneous

masso

nbo

thT1-,T2

-and

T1-w

eighted

imagea

fterg

adolinium

injection;

cysticchangesw

/flu

idlevels

Nezhat,2005

(1)

3-cm

hepatic

cystin

thefar

caud

alaspect

ofther

ight

lobe

oftheliver

3-cm

hepatic

cystin

thefar

caud

alaspect

ofther

ight

lobe

oftheliver

N/A

Nezhat,2005

(2)

Normalfin

ding

sN/A

Normalfin

ding

s

Reid,2003

10cm

massw

ithecho

genicm

argins

and

internaldebris

Lowdensity

lesio

nN/A

Rivkine,2013

80x75

mm

intraparenchym

alhepatic

necrotictumor

Hypovasculariz

ed,cystic

massintheL

liver

lobe

with

hemorrhagiccontents,

noseptations

Cysticm

assinsegm

entsIIandIII

Case Reports in Hepatology 7

Table2:Con

tinued.

Author,Year

US

CTMRI

Roesch-D

ietlan,

2011

Nomasses,multip

lesm

allgallston

esN/A

N/A

Rovati,

1990

10cm

cysticmassw

ithseptations

Multilocular

10cm

cystwith

fine

calcificatio

nsin

thew

all

N/A

Schu

ld,2011

N/A

N/A

6.8x2.3cm

indiam

eter

inther

ight

basallun

gand

perip

heralbile

ducts

Sherif,

2016

3cm

complex

cystin

Rlobes

3cm

welld

efinedhypo

denses

ubcapsular

lesio

nin

Rlobe

with

heterogeneou

sperip

heralenh

ancementinthev

enou

sanddelayedph

ases

Subcapsularp

artia

llycysticfocallesionwith

possible

hemorrhagiccontentand

heterogeneou

speripheral

enhancem

ent

Tuech,2003

N/A

24cm

smoo

thwalledcysticlesionwith

out

septations

intheR

lobe

N/A

Verbeke,1996

(1)

N/A

Cystictumor

inRlobe

ofliver,w

ithreactiv

eenlargemento

fLhepatic

lobe

Cystictumor

inRlobe

ofliver,w

ithreactiv

eenlargem

ento

fLhepatic

lobe

Verbeke,1996

(2)

Cyst(12x10

x7.5

cm)intheleft

liver

lobe,located

near

theg

allbladd

erandthe

liver

hilus,which

partially

compressed

thep

roximaldu

ctus

choledochu

s.

Cyst(12x10

x7.5

cm)intheleft

liver

lobe,located

near

theg

allbladd

erandthe

liver

hilus,which

partially

compressed

thep

roximaldu

ctus

choledochu

s.

N/A

Rana

2019

N/A

3.2cm

x4cm

x1.8

cmmulti-septated

cysticlesio

nin

theleft

hepatic

lobe

N/A

8 Case Reports in Hepatology

Authors’ Contributions

Prachi Rana and Shida Haghighat equally contributed to thecollection of the data and writing of the manuscript. HyosunHan reviewed and edited the manuscript.

Acknowledgments

We thank Dr. Yangling Ma and Dr. Esteban Gnass for thereview and interpretation of the pathologic reports.

References

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