endometrial carcinoma in a gravid uterus: a case report and ......case report open access...

CASE REPORT Open Access

Endometrial carcinoma in a gravid uterus: acase report and literature reviewMayu Shiomi, Shinya Matsuzaki* , Eiji Kobayashi, Takeya Hara, Satoshi Nakagawa, Tsuyoshi Takiuchi,Kazuya Mimura, Yutaka Ueda, Takuji Tomimatsu and Tadashi Kimura

Abstract

Background: Endometrial carcinoma (EC) is rarely diagnosed during pregnancy. Therefore, the histopathologicalfindings, clinical course, and gross appearance of the resected uterus during pregnancy are not well known. Wepresent a case of EC diagnosed during pregnancy. In addition, we reviewed the literature dating from January 1995to March 2019 for cases of EC diagnosed during pregnancy and within 15months after pregnancy, and we discussedthis topic to improve the understanding of this rare condition.

Case presentation: A 35-year-old woman underwent an urgent cesarean delivery in gestational week 35 due toantepartum bleeding caused by placenta previa. Hysterectomy was performed with the diagnosis of placenta accretaspectrum (PAS). Remarkably, the postoperative gross and histopathological examinations revealed an endometrioidadenocarcinoma (grade 1). The histopathological findings revealed a pattern similar to that of EC not related withpregnancy. Immunohistochemistry revealed an overexpression of the estrogen and progesterone receptors; however,the p53 expression was negative. We performed laparoscopic bilateral salpingo-oophorectomy and pelviclymphadenectomy 102 days after the cesarean hysterectomy, and confirmed surgical stage IA without metastases. Ourpatient has had no recurrence in 4 years after the cesarean delivery.An electronic search of the literature revealed 25 cases of EC (including our case) diagnosed during or after pregnancy.Sixteen of the 25 patients were diagnosed after abortions in the first trimester, 9 were diagnosed within 14months ofchildbirth, and our case was the first with diagnosis from a surgical specimen of peripartum hysterectomy due to thePAS. In 23 of the 25 cases endometrioid adenocarcinoma grade 1 to 2 was found, and it seemed to have a goodprognosis.

Conclusion: The present findings suggest that careful examination of a resected uterus is essential, even when surgeryis performed for an obstetric indication. Our case is an extremely rare case of EC during pregnancy; the histopathologicalpattern was similar to that of typical EC, and no recurrence was noted. The high levels of estrogen and progesteroneduring pregnancy did not seem to promote tumor progression in our case.

Keywords: Placenta accreta spectrum, Placenta previa, Pregnancy, Endometrioid carcinoma endometrial carcinoma,Endometrial cancer

This study presents a case of endometrioid carcinomadiagnosed during pregnancy. We performed literaturereview and discussed this topic. We have discussed theeffects of pregnancy on endometrioid carcinoma in aprevious study. Our present study found the points listedbelow.

1. Although endometrial carcinoma during pregnancyis extremely rare, careful observation of the resecteduterus is needed to avoid a missed diagnosis.

2. In our case, histopathological andimmunohistochemical findings were consistent withendometrioid adenocarcinoma grade 1. The patienthas been disease-free for about 4 years aftercesarean hysterectomy. The high levels of estrogenand progesterone during pregnancy did not seem topromote tumor progression in our case.

© The Author(s). 2019 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

* Correspondence: [email protected] of Obstetrics and Gynecology, Osaka University Graduate Schoolof Medicine, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan

Shiomi et al. BMC Pregnancy and Childbirth (2019) 19:425 https://doi.org/10.1186/s12884-019-2489-y

http://crossmark.crossref.org/dialog/?doi=10.1186/s12884-019-2489-y&domain=pdfhttp://orcid.org/0000-0001-5725-9994http://creativecommons.org/licenses/by/4.0/http://creativecommons.org/publicdomain/zero/1.0/mailto:[email protected]

3. Although high levels of estrogen (which has apromoting effect on endometrioid carcinoma) andprogesterone (which has an anti-tumor effect onendometrioid carcinoma) were observed, mostauthors reported that the endometrioid carcinomaassociated with pregnancy had a good prognosiswith minimal myometrial invasion.

BackgroundEndometrial carcinoma (EC) is the fourth most commoncancer in women in high-income countries; however, ECcommonly occurs in peri- or postmenopausal women,and only 5% of women are diagnosed with adenocarcin-oma before the age of 40 years [1, 2]. Therefore, thecoexistence of EC and pregnancy is rare. Moreover, ECis rarely detected during pregnancy or within a yearpostpartum because the tumor can disrupt the preg-nancy. Although a previous study had already reviewedthe latest 35 reports on EC coexisting with pregnancyduring the last 80 years [3], the outcome of EC associ-ated with pregnancy and the effect of pregnancy on ECis not well known. Previous literature review alsoshowed that there have been no reports of diagnosingEC during pregnancy in the surgical specimen ofcesarean hysterectomy. Therefore, we report a case ofEC diagnosed in a postoperative histopathological exam-ination after total hysterectomy for placenta accretaspectrum (PAS), and we additionally present the resultsof a literature review on this matter.

Case presentationA 35-year-old woman (gravida 2, para 1) was referred toour hospital due to placenta previa at gestational week31. Her medical history was unremarkable, and her pre-vious pregnancy was an uncomplicated, normal vaginal

delivery at gestational week 38. Her current pregnancywas uncomplicated except for the placenta previa. Shedenied abnormal genital bleeding before the currentpregnancy. Cervical cytology performed during earlypregnancy was negative for intraepithelial lesions. Vagi-nal ultrasonography revealed total placenta previa andone lacuna (Fig. 1a). Magnetic resonance imaging (MRI)at gestational week 31 revealed total placenta previa andloss of the myometrium between the placenta and blad-der wall (Fig. 1b). Other MRI findings of PAS such asuterine bulging, heterogenous placenta, and T2 darkband were not observed. Based on these findings, wesuspected PAS, and an emergency cesarean delivery wasperformed owing to antepartum bleeding (approximately100 mL) at gestational week 35. An abdominal midlineincision was made, and a healthy male infant weighing2274 g (− 0.42 SD) was delivered with Apgar scores of 8and 9, at 1 and 5min, respectively. The placenta was notdelivered within 30min after fetal delivery, thus requir-ing hysterectomy for PAS. Estimated blood loss was1000 mL. The postoperative course was uneventful, andthe patient and baby were discharged on the 8th postop-erative day.Part of the chorion and placenta were adhered to the

uterus (Fig. 2a). The resected uterus was divided to 7specimens in order to perform macroscopic and histo-pathological analyses. The surgical specimen showed awhite polyp measuring 2 cm, which parted from theuterine fundus and the lower uterine segment (Fig. 2b).Histopathological examination of the tumor involvingthe lower uterine segment revealed endometrioid adeno-carcinoma (Grade 1), with < 50% myometrial invasionand positive expression of estrogen and progesterone re-ceptors, in addition to PAS (Fig. 3a and b). Notably, thetumor involving the uterine fundus did not show

Fig. 1 Images for assessment of placenta accreta spectrum. a Transvaginal ultrasonography shows total placenta previa with one lacuna. bMagnetic resonance imaging (MRI) at gestational week 31 revealed total placenta previa, and the placenta was located mainly on the anteriorside. Although intraplacental T2 dark band, uterine bulging, and heterogeneous placenta were not observed, we found myometrial thinning ofthe anterior wall and loss of myometrium between the placenta and bladder wall. The black arrow indicates loss of uterine myometriumbetween the placenta and bladder wall. Based on these findings, we suspected placenta accreta spectrum. No abnormal finding was observed inthe fetus

Shiomi et al. BMC Pregnancy and Childbirth (2019) 19:425 Page 2 of 9

myometrial invasion. Histopathological findings weresimilar in both tumors located in the uterine lower seg-ment and uterine fundus. A retrospective review of theMRI images obtained during pregnancy revealed thetumor involving the uterine fundus, although involvementof the lower uterine segment was difficult to detect(Fig. 3c). We performed a laparoscopic bilateral salpingo-oophorectomy and pelvic lymphadenectomy 102 daysafter cesarean hysterectomy and confirmed the absence ofmetastases. The tumor was a stage IA lesion based on theInternational Federation of Gynecology and Obstetricssystem. Follow-up performed 4 years after cesareanhysterectomy revealed no recurrence.

Discussion and conclusionOur case demonstrated the gross and histopathologicalfindings, MRI findings, and clinical course of EC duringpregnancy. To discuss this rare condition, we performeda literature review of cases of endometrioid carcinomaassociated with pregnancy. We defined EC associatedwith pregnancy as diagnosed at delivery to within 15months after pregnancy. We performed a search ofPubMed, MEDLINE, and Scopus databases for theperiod between January 1995 and March 2019, using thefollowing key words: “endometrial cancer”, “endometrialcarcinoma”, “endometrioid cancer”, “endometrioid car-cinoma”, “corpus cancer”, “pregnancy”, “abortion”, and“postpartum” in various combinations. We excludednon-English articles, discontinued journal and thosepublished before 1995. We summarized the timing ofdiagnosis, outcome of EC, symptoms, diagnosis of histo-pathological examination, surgical stage (base on FIGO

2008) [4], and surgical treatment for EC. We also listedthe authors’ opinions and discussions about the effect ofpregnancy on the prognosis of EC.A total of 18 studies with 25 cases of EC associated

with pregnancy (including our case) have been reported[3, 5–20]; 9 cases were identified postpartum up to the14-month; 16 cases were diagnosed at the time of D&Cfor first-trimester spontaneous or elective abortion.These results suggest that clinicians should consider ECafter pregnancy even though abnormal bleeding is oftenobserved after pregnancy, and EC associated with preg-nancy is rare. Our literature review revealed that therewere no previous reports of a diagnosis of EC based onan examination of the resected uterus following cesareanhysterectomy for PAS [3, 7]. Although our case is ex-tremely rare, clinicians should check the macroscopicfinding of the resected uterus carefully regardless of theindication for hysterectomy.Our literature review showed that the histopatho-

logical classification was endometrioid adenocarcinomagrade 1–2 in 23 of the 25 cases, unknown grade of endo-metrioid adenocarcinoma in 1 of the 25 cases, andpoorly differentiated adenosquamous carcinoma in 1 ofthe 25 cases. Immunohistochemical (IHC) analysis wasperformed in 9 of the 24 cases and revealed a typicalstaining pattern as previously reported [21, 22]. Previousreports have shown that women younger than 45 yearsrarely developed EC, and the most common subtype ofclassification in younger women was endometrioidadenocarcinoma grade 1–2 [23, 24]. Although the num-ber was limited, these results suggested that pregnancydid not affect the subtype and IHC staining pattern of

Fig. 2 Macroscopic findings in the surgical specimen. a The image shows gross findings in the uterus, which was resected due to placentaaccreta spectrum. The white arrow indicates a white tumor measuring 3 cm in diameter, involving the lower uterine segment, which wasdiagnosed as endometrial carcinoma by histopathological analysis. The tumor involving the uterine fundus is not identifiable because it iscovered by the placenta. b The image shows a longitudinal section of the uterus, which was divided into 7 sections. After the placenta wasremoved, a white tumor measuring 2 cm in diameter involving the uterine fundal segment was seen. The black arrow indicates the 3-cmdiameter tumor which was endometrial carcinoma involving the lower uterine segment; the white arrow indicates the tumor involving theuterine fundus. Both tumors were soft and white, and the macroscopic findings were similar in both tumors


EC. We considered that our literature review might bebiased because we could include only published litera-ture and cases that made it to the scientific publicationstage and this condition might be under-reported; thus,this is the limitation of our study.The case we presented is rare, and this report high-

lights several interesting points, as follows: 1. It describesthe histopathological analysis of EC during pregnancy; 2.It describes a tumor involving the lower uterine segmentand simultaneously the uterine fundus; and 3. Itdescribes the MRI appearance of EC during pregnancy.Histopathological examination of the specimen re-

vealed EC that presented as a well-differentiated adeno-carcinoma with a focal cribriform pattern, back-to-backstructure, and a papillary area. Although IHC analysisshowed positive expression of estrogen and progesteronereceptors, our patient did not demonstrate any metasta-ses, and no recurrence was observed 4 years after thecesarean hysterectomy. These features resemble those of

typical grade 1 endometrioid adenocarcinoma [25–27].We concluded that the high-dose estrogen and proges-terone condition during pregnancy did not promote pro-gression of the EC. As shown in Table 1, most authorsconsidered that pregnancy did not worsen the prognosisof EC. Further cases are expected to discuss how thepregnancy affects the prognosis of EC. Moreover, thehistopathological and IHC findings in our case showedsimilar pattern to those of typical EC.The reason for the presentation of separate tumors at

the uterine fundus and lower uterine segment isunknown. Histopathological analysis of both tumorsshowed similar findings; thus, we concluded that thetumor presented as 2 separate growths at the aforemen-tioned sites owing to enlargement of the uterus duringpregnancy (although this remains speculative). Otherpossibilities considered were metastasis or multi-site in-volvement of EC. Myometrial invasion was insignificant;thus, we excluded metastasis as a possible etiology. The

Fig. 3 Postoperative analysis of histopathological findings, magnetic resonance imaging, and immunohistochemistry staining. a The image showsthe histopathological findings in the resected uterine specimen. Well-differentiated adenocarcinoma with focal cribriform pattern, back-to-backstructure without intervening stroma, and a papillary area are observed, and the glands have a smooth luminal contour. The tumor showspredominant glandular growth and a < 5% nonsquamous solid component; thus, the tumor was diagnosed as endometrial cancer grade 1. Thetumor at the lower uterine segment shows slight myometrial invasion. The white arrow indicates the tumor in the uterine lower segment whichshows invasion of the placenta decidua and uterine myometrium. The black arrow indicates < 50% myometrial invasion (hematoxylin and eosinstain, × 40.) b Immunohistochemistry analysis showed positive expression of estrogen and progesterone receptors, and negative expression ofp53. (Magnification, × 40.) c Retrospectively reviewed magnetic resonance imaging (MRI) revealed endometrial carcinoma in the uterine fundus. Asagittal T2-weighted MR image shows endometrial carcinoma measuring 3 cm in diameter with signal intensity resembling that of the placenta.The white arrow indicates endometrial carcinoma involving the uterine fundus


Table

1Asummaryof

theliteraturereview

finding

sforen

dometrio

idcarcinom

aassociated

with

preg

nancy

Firstauthor

Year

(Referen

cenu

mbe

r)

Age

(years)

Timingof

diagno

sis

Outocom

ePerio

dafter

diagno

sis

Symptom

sTheresults

ofhistop

atho

gical

exam

ination

Immun

ohisotoche

ical

staining

Stage

Surgicaltreatm

ent

Kovács

AG

1996

[5]

35Abo

rtion

NA

NED

1year

Abn

ormalge

nital

bleeding

EAgrade1–2

NA

IABrachytherapy+TA

H+BSO+RT

Theauthorshypo

thesized

that

preg

nancymay

adverselyaffect

thetumor

grow

th;how

ever,itcann

otbe

proven

becauseof

thelim

itednu

mbe

rof

cases.

KodamaJ

1997

[6]

30Po

stpartum

7mon

ths

DOD

8 mon

ths

Abn

ormalge

nital

bleeding

Poorlydifferentiated

aden

osqu

amou

scarcinom

a

NA

IIIC

C

Theauthorsop

ined

that

anim

mature,prog

esterone

-unrespo

nsiveen

dometriu

mcouldbe

thepo

ssiblemechanism

ofallowingen

dometrialcarcino

mato

developin

preg

nancy.

Schammel

DP

1998

[7]

38Abo

rtion

9weeks

NED

58 mon

ths

Infertility

EAgrade1

NA

IARepe

atcurettagewith

prog

esterone

therapy

41Abo

rtion

13weeks

NED

48 mon

ths

Abn

ormalge

nital

bleeding

EAgrade1

NA

IATA

H+BSO

29Abo

rtion

9–10

weeks

NA

NA

Non

eEA

grade1

NA

IANA

34Abo

rtion

13weeks

NED

12 mon

ths

Abn

ormalge

nital

bleeding

EAgrade1

NA

IATA

H+BSO

33Po

stpartum

During

cesarean

delivery

NED

57 mon

ths

Non

eEA

grade1

NA

IARepe

atcurettagewith

prog

esterone

therapy

Theauthorsconsidered

that

thefate

ofthemoreadvanced

-stage

tumorswith

deep

ermyometrialinvasionor

high

-grade

cytologicfeatures

may

beless

subjectto

theprotectiveeffectsof

gestational

prog

esterone

.

Ayhan

A1999

[8]

44Abo

rtion

5weeks

NA

NA

Abn

ormalge

nital

bleeding

EAgrade1

NA

IATA

H+BSO+LN

D+OM

Theauthorscitedaprevious

repo

rtwhich

observed

that

hCGinhibitstheDMBA

-indu

cedbreastcarcinog

enesisin

ratsthroug

han

insulin-like

grow

thfactor-dep

ende

ntmechanism

.

Foersterling

DL

1999

[9]

31Po

stpartum

9weeks

NED

1year

Abn

ormalge

nital

bleeding

EAgrade1

NA

IATA

H+BSO

Theauthorsop

ined

that

inpreg

nancy-associated

endo

metrialcarcino

ma,partof

theliningun

dergoe

sge

stationalchang

e,whe

reas

anothe

rpartbe

comes

neop

lastic.The

portionof

theen

dometriu

mwhich

becomes

neop

lasticmay

besensitive

toestrog

en,yet

unrespon

sive

toprog

esterone

.

Vaccarello

L1999

[10]

35Abo

rtion

9weeks

NED

31 mon

ths

Abn

ormalge

nital

bleeding

EAgrade1

NA

IATA

H+BSO

40Po

stpartum

4mon

ths

NED

6years

Abn

ormalge

nital

bleeding

EAgrade1

NA

IATA

H+BSO

32Po

stpartum

4mon

ths

NED

3.5years

Abn

ormalge

nital

bleeding

EAgrade2

NA

NA

TAH+BSO

They

conclude

dthat

with

concom

itant

secretoryen

dometriu

m,the

malignant

region

smustbe

prog

esterone

refractory.

Mitsushita

J2000

[11]

28Po

stpartum

6mon

ths

NA

NA

Previous

historyof

endo

metrio

idcarcinom

a

EAgrade1

ER:p

ositive

PR:p

ositive

IATA

H

Theauthorsdidno

tdiscusstheassociationbe

tweenpreg

nancyanden

dometrio

idcarcinom

a.


Table

1Asummaryof

theliteraturereview

finding

sforen

dometrio

idcarcinom

aassociated

with

preg

nancy(Con

tinued)

Firstauthor

Year

(Referen

cenu

mbe

r)

Age

(years)

Timingof

diagno

sis

Outocom

ePerio

dafter

diagno

sis

Symptom

sTheresults

ofhistop

atho

gical

exam

ination

Immun

ohisotoche

ical

staining

Stage

Surgicaltreatm

ent

Kovács

AG

1996

[5]

35Abo

rtion

NA

NED

1year

Abn

ormalge

nital

bleeding

EAgrade1–2

NA

IABrachytherapy+TA

H+BSO+RT

IshiokaS

2000

[12]

25Po

stpartum

14mon

ths

NED

6 mon

ths

Abn

ormalge

nital

bleeding

EAgrade1

ER:p

ositive

PR:neg

ative

p53:ne

gative

IAmRH

+BSO+LN

D

Theauthorsconclude

dthat

theoccurren

ceof

postpartum

ECwas

extrem

elyrare

prob

ablydu

eto

theanti-tumor

effectsof

prog

esterone

.

IchikawaY

2001

[13]

35Po

stpartum

6mon

ths

NED

3.5years

Lower

abdo

minal

pain

EAgrade1

NA

IBTA

H+BSO+LN

D+OM+

App

ende

ctom

y

Theauthorsspeculated

that

high

prog

esterone

levelsdu

ringpreg

nancymay

protectagainstEC

.

ItohK

2004

[14]

39Po

stpartum

6mon

ths

NED

3years

Abn

ormalge

nital

bleeding

EAgrade1

ER:neg

ative

PR:neg

ative

IBTA

H+BSO+LN

D

Theauthorsconclude

dthat

theanticancereffect

ofprog

esterone

durin

gpreg

nancywas

ineffect

inthesetumors.

Hannu

naKY

2009

[3]

34Abo

rtion

12weeks

NED

18 mon

ths

Abo

rtion

EAgrade1–2

ER:p

ositive

PR:p

ositive

CK7:p

ositive

CK20:ne

gative

β-hC

G:neg

ative

E-cadh

erin:p

ositive

EpCAM:p

ositive

Placen

talalkaline

phosph

atase:po

sitive

IAD&C

Theauthorsspeculated

that

thepresen

ceof

ECmight

have

been

relatedto

ahypo

xicdamageof

thechorionicvilli.Itmight

sugg

estacausalcorrelationbe

tweenen

dometrialm

alignancyand

spon

tane

ousabortio

n.

Theauthorsfoun

dthat

mostcase

repo

rtsof

firsttrim

esterEA

arealso

repo

rted

asarisingin

afocallesion.

Terada

T2009

[15]

29Con

curren

ten

dometrial

aden

ocarcino

maandan

early

preg

nancyloss

NA

NA

Abo

rtion

EAgrade2

ER:p

ositive

PR:p

ositive

p53:po

sitive

vimen

tin:positive

CA19–9:focalpo

sitive

CA125:po

sitive

Ki-67:80%

labe

lling

CEA

:neg

ative

PTEN

:neg

ative

p16:ne

gative

NA

Repe

atcurettagewith

out

prog

esterone

therapy


that

ECassociated

with

preg

nancyweremostly

instages

IA,and

werehistolog

icallyEA

s.

AkilA

2012

[16]

45Con

curren

ten

dometrial

aden

ocarcino

maandan

early

preg

nancyloss

NA

NA

Abo

rtion

EAgrade1

NA

IATA

H+BSO+LN

D

Theauthorsconclude

dthat

theroutinehistolog

icalexam

inationof

thecurettagespecim

ensforallfirsttrim

esterabortio

ns,ind

epen

dent

oftheageof

thepatient,sho

uldbe

encouraged

.

SaciragicL

2014

[17]

36Abo

rtion

8weeks

NA

NA

Abn

ormalge

nital

bleeding

EAgrade1

Ki67:p

ositive

IATA

H+BSO+LN

D


Table

1Asummaryof

theliteraturereview

finding

sforen

dometrio

idcarcinom

aassociated

with

preg

nancy(Con

tinued)

Firstauthor

Year

(Referen

cenu

mbe

r)

Age

(years)

Timingof

diagno

sis

Outocom

ePerio

dafter

diagno

sis

Symptom

sTheresults

ofhistop

atho

gical

exam

ination

Immun

ohisotoche

ical

staining

Stage

Surgicaltreatm

ent

Kovács

AG

1996

[5]

35Abo

rtion

NA

NED

1year

Abn

ormalge

nital

bleeding

EAgrade1–2

NA

IABrachytherapy+TA

H+BSO+RT

Theauthorsdiscussedthat

inawom

anwith

prog

esterone

-resistant

endo

metriu

m,d

evelop

men

tof

endo

metrialcarcino

macouldbe

potentiatedby

therelativelyhype

restroge

nicen

vironm

entof

early

preg

nancyandsubseq

uentlyallowed

toproliferate

furthe

rdu

eto

alack

ofrespon

seto

prog

esterone

.

Bayoglu

TekinY

2014

[18]

36Abo

rtionor

ectopic

preg

nancy

NA

NED

1year

Ectopicpreg

nancy

EAgrade1

NA

NA

Curettage

with

prog

esterone

therapy

Theauthorsthou

ghthat

thepresen

ceof

ECmight

have

been

relatedto

thedamageof

thechorionicvilli,sug

gestingacausalcorrelationbe

tweenEC

andspon

tane

ousabortio

ns.

Zhou

F2015

[19]

40Con

curren

ten

dometrial

aden

ocarcino

maandan

early

preg

nancyloss

NA

NA

Abo

rtion

EAgrade1

ER:p

ositive

PR:p

ositive

p53:ne

gative

NA

Repe

atcurettagewith

out

prog

esterone

therapy

33Con

curren

ten

dometrial

aden

ocarcino

maandan

early

preg

nancyloss

NA

NA

Abo

rtion

EAgrade1

ER:p

ositive

PR:p

ositive

p53:ne

gative

NA

Repe

atcurettagewith

out

prog

esterone

therapy


that

thecarefulh

istologicalexaminationof

thecurettagespecim

ensforallfirsttrim

esterpreg

nancylosses

shou

ldbe

encouraged

.

RizzutoI

2019

[20]

29Preg

nancyof

7weeks

gestation

NA

NED

8years

Abn

ormalge

nital

bleeding

EANA

NA

Serialend

ometrialb

iopsywith

insertionof

aLevono

rgestrel

intrauterin

ede

vice

Con

servativemanagem

entforEC

inyoun

gwom

enispo

ssibleinclud

ingacase

with

anincide

ntaldiagno

sisin

preg

nancy.

Our

case

2019

35Placen

taaccreta

spectrum

Cesarean

hysterectomy

NED

4years

Non

eEA

grade1

ER:p

ositive

PR:p

ositive

p53:ne

gative

IACesareanhysterectomy

Laparoscop

icBSO+LN

D

List

ofab

breviatio

ns:B

SOBilateralsalping

o-oo

phorectomy,CChe

mothe

rapy

,CK7

Cytok

eratin

7,CK

20Cytok

eratin

20,C

A19–9

Can

ceran

tigen

19–9

,CA125Can

ceran

tigen

125,

CEACarcino

embryo

nican

tigen

,D&C

Dilatatio

nan

dcurettag

e,DODDeadof

disease,

EAEA

,ECen

dometrio

idcarcinom

a,EpCA

MEp

ithelialcella

dhesionmolecule,

EREstrog

enreceptor,β

-hCG

Hum

anchorionicgo

nado

trop

inβ-subu

nit,LN

DLymph

node

dissectio

n,mRH

Mod

ified

radicalh

ysterectom

y,NANot

available,

NED

Noeviden

ceof

disease,

OM

Omen

tectom

y,PR

Prog

esterone

receptor,R

TRa

diationtherap

y,TA

HTran

sabd

ominal

hysterectomy


possibility of multi-site involvement of EC is difficult toexclude; however, the estimated frequency of this condi-tion is low. Therefore, we concluded that the tumor sep-aration could be attributed to the uterine enlargementduring pregnancy.MRI scans were retrospectively analyzed after the

cesarean hysterectomy. We observed a lesion in theuterine fundus measuring approximately 3 cm in diam-eter with signal intensity similar to that of the placenta.Notably, this lesion was separate from the placenta. Cli-nicians must consider the possibility of EC in womenwith MRI scans showing such lesions during pregnancy.In conclusion, our findings in this case suggest that

careful analysis of MRI findings during pregnancy andgross examination of the resected uterus (in patientsundergoing hysterectomy for obstetric complications)are essential, although EC during pregnancy is extremelyrare. The literature review suggested that EC associatedwith pregnancy seemed to have a good prognosis.

AbbreviationsD&C: Dilatation & Curettage; EC: Endometrial carcinoma;IHC: Immunohistochemical; MRI: Magnetic resonance imaging; PAS: Placentaaccreta spectrum

AcknowledgementsThe authors thank H. Abe and K. Sakiyama for administrative assistance inthe preparation of this manuscript.

Authors’ contributionsMS, SM, EK, and YU made substantial contributions to conception anddesign, collected the clinical data and drafted as well as revised themanuscript. EK, TH, SN, TTa, TTo, and KM helped in reviewing the previousstudies and drafting the manuscript. TK conceived and generally supervisedof this study, and gave final approval of the version to be published. Allauthors read and approved the final manuscript.

FundingThere is no source of financial support or funding.

Availability of data and materialsNot applicable.

Ethics approval and consent to participateThis study was approved by the Institutional Review Board and the EthicsCommittee of the Osaka University Hospital (approval #15240, approved onSeptember 10, 2015).

Consent for publicationWritten informed consent was obtained from the patient for publication ofthis case report and any accompanying images. A copy of the writtenconsent is available for review by the Editor of this journal.

Competing interestsShinya Matsuzaki is an Associate Editor for BMC Pregnancy and Childbirth.The authors declare no conflicts of interest about this study. All of authorshave no competing financial interests regarding this study.

Received: 3 June 2019 Accepted: 4 September 2019

References1. Soliman PT, Oh JC, Schmeler KM, Sun CC, Slomovitz BM, Gershenson DM,

et al. Risk factors for young premenopausal women with endometrialcancer. Obstet Gynecol. 2005;105:575–80.

2. Jordan SJ, Na R, Johnatty SE, Wise LA, Adami HO, Brinton LA, et al.Breastfeeding and endometrial Cancer risk: an analysis from theepidemiology of endometrial Cancer consortium. Obstet Gynecol. 2017;129:1059–67.

3. Hannuna KY, Putignani L, Silvestri E, Pisa R, Angioli R, Signore F. Incidentalendometrial adenocarcinoma in early pregnancy: a case report and reviewof the literature. Int J Gynecol Cancer. 2009;19:1580–4.

4. Pecorelli S. Revised FIGO staging for carcinoma of the vulva, cervix, andendometrium. Int J Gynaecol Obstet. 2009;105:103–4.

5. Kovacs AG, Cserni G. Endometrial adenocarcinoma in early pregnancy.Gynecol Obstet Investig. 1996;41:70–2.

6. Kodama J, Yoshinouchi M, Miyagi Y, Kobashi Y, Kamimura S, Okuda H, et al.Advanced endometrial cancer detected at 7 months after childbirth.Gynecol Oncol. 1997;64:501–6.

7. Schammel DP, Mittal KR, Kaplan K, Deligdisch L, Tavassoli FA. Endometrialadenocarcinoma associated with intrauterine pregnancy. A report of fivecases and a review of the literature. Int J Gynecol Pathol. 1998;17:327–35.

8. Ayhan A, Gunalp S, Karaer C, Gokoz A, Oz U. Endometrial adenocarcinomain pregnancy. Gynecol Oncol. 1999;75:298–9.

9. Foersterling DL, Blythe JG. Ovarian carcinoma, endometrial carcinoma, andpregnancy. Gynecol Oncol. 1999;72:425–6.

10. Vaccarello L, Apte SM, Copeland LJ, Boutselis JG, Rubin SC. Endometrialcarcinoma associated with pregnancy: a report of three cases and review ofthe literature. Gynecol Oncol. 1999;74:118–22.

11. Mitsushita J, Toki T, Kato K, Fujii S, Konishi I. Endometrial carcinomaremaining after term pregnancy following conservative treatment withmedroxyprogesterone acetate. Gynecol Oncol. 2000;79:129–32.

12. Ishioka S, Sagae S, Saito T, Sugimura M, Akutagawa N, Nishimura M, et al. Acase of uterine endometrial carcinoma 15 months post-partum. J ObstetGynaecol Res. 2000;26:417–20.

13. Ichikawa Y, Takano K, Higa S, Tanabe M, Wada A, Sugita M, et al.Endometrial carcinoma coexisting with pregnancy, presumed to derivefrom adenomyosis: a case report. Int J Gynecol Cancer. 2001;11:488–90.

14. Itoh K, Shiozawa T, Shiohara S, Ashida T, Konishi I. Endometrial carcinoma inseptate uterus detected 6 months after full-term delivery: case report andreview of the literature. Gynecol Oncol. 2004;93:242–7.

15. Terada T. Endometrial adenocarcinoma in spontaneous abortion. Pathology.2009;41:694–5.

16. Akil A, Kaya B, Karabay A, Kibar Y. Concurrent endometrial adenocarcinomaand an early pregnancy loss. Arch Gynecol Obstet. 2012;286:1089–90.

17. Saciragic L, Ball CG, Islam S, Fung-Kee-Fung M. Incidental endometrialcarcinoma diagnosed at first trimester pregnancy loss: a case report. JObstet Gynaecol Can. 2014;36:1010–3.

18. Bayoglu Tekin Y, Guvendag Guven ES, Sehitoglu I, Guven S. Tubalpregnancy associated with endometrial carcinoma after in vitro fertilizationattempts. Case Rep Obstet Gynecol. 2014;2014:481380.

19. Zhou F, Qian Z, Li Y, Qin J, Huang L. Endometrial adenocarcinoma inspontaneous abortion: two cases and review of the literature. Int J Clin ExpMed. 2015;8:8230–3.

20. Rizzuto I, Nicholson R, Dickinson K, Juang HJ, MacNab W, Rufford B. A caseof incidental endometrial adenocarcinoma diagnosed in early pregnancyand managed conservatively. Gynecol Oncol Rep. 2019;28:101–3.

21. Cancer Genome Atlas Research N, Kandoth C, Schultz N, Cherniack AD,Akbani R, Liu Y, et al. Integrated genomic characterization of endometrialcarcinoma. Nature. 2013;497(7447):67–73.

22. Murali R, Soslow RA, Weigelt B. Classification of endometrial carcinoma:more than two types. Lancet Oncol. 2014;15:e268–78.

23. Reed SD, Newton KM, Clinton WL, Epplein M, Garcia R, Allison K, et al.Incidence of endometrial hyperplasia. Am J Obstet Gynecol. 2009;200:678 e1–6.

24. Committee on Practice B-G. Practice bulletin no. 128: diagnosis of abnormaluterine bleeding in reproductive-aged women. Obstet Gynecol. 2012;120:197–206.

25. Matsuo K, Machida H, Shoupe D, Melamed A, Muderspach LI, Roman LD,et al. Ovarian conservation and overall survival in young women with early-stage low-grade endometrial Cancer. Obstet Gynecol. 2016;128:761–70.

26. Sorosky JI. Endometrial cancer. Obstet Gynecol. 2008;111(2 Pt 1):436–47.27. Guan J, Xie L, Luo X, Yang B, Zhang H, Zhu Q, et al. The prognostic

significance of estrogen and progesterone receptors in grade I and IIendometrioid endometrial adenocarcinoma: hormone receptors in riskstratification. J Gynecol Oncol. 2019;30:e13.


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AbstractBackgroundCase presentationConclusion

BackgroundCase presentationDiscussion and conclusionAbbreviationsAcknowledgementsAuthors’ contributionsFundingAvailability of data and materialsEthics approval and consent to participateConsent for publicationCompeting interestsReferencesPublisher’s Note

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