6
Oman Medical Specialty Board Endometriosis After Surgical Menopause Mimicking Pelvic Malignancy: Surgeons’ Predicament Rani A. Bhat, Melissa Teo and Akhil Krishnanand Bhat Rani A. Bhat Consultant Gynaecological Oncologist HCG Oncology No. 8, P Kalinga Rao Road Sampangi Ramnagar Bangalore - 560027. India. E-mail: [email protected] Melissa Teo Senior Consultant, Department of Surgical Oncology, National Cancer Centre Singapore, Singapore. Akhil Krishnanand Bhat Senior Consultant-General Surgery, St. Philomena's Hospital, No. 4 Cambell Road, Viveknagar, Bangalore-560047, India. Abstract Prevalence of persistent endometriosis in women after menopause without any hormonal replacement therapy is very rare. is is a case of a woman with previous history of total hysterectomy and bilateral salpingo-oophorectomy for endometriosis who presented with hemoperitoneum, vaginal bleeding, pelvic mass, and pulmonary thromboembolism mimicking as rectovaginal septum carcinoma. is is the first case report with a unique mode of presentation wherein the patient presented with hemoperitoneum requiring emergency embolization of the vessel to stabilize the patient. She underwent en bloc resection of the tumor with high anterior resection of the rectum. Histopathology confirmed endometriosis. Keywords: Endometriosis; Ovarian cancer; Menopause. Introduction The incidence of endometriosis in post-menopausal women is 2% to 5% 1,2 and is commonly seen in those who have received hormonal replacement therapy after menopause. 3 is report is about a rare case of a patient with post-menopausal endometriosis, who presented with intra-abdominal bleeding, pelvic mass, vaginal bleeding and pulmonary thromboembolism. With clinical and radiological findings and past history of endometriosis, the disease was considered as rectovaginal septum carcinoma of clear cell type resulting from malignant transformation of endometriosis. Received: 10 Feb 2014 / Accepted: 12 Apr 2014 © OMSB, 2014 Case Report A 50-year-old para 2 was referred to our hospital in view of pelvic mass, bilateral hydronephrosis and pulmonary embolism. She had initially presented to a private hospital with a history of acute abdomen due to hemoperitoneum and bleeding per-vagina. A contrast enhanced CT scan of the abdomen and pelvis showed large hemoperitoneum with solid dense areas in the caudal portion of the pelvis with a blush of increasing contrast enhancement in the central part of the pelvis indicating active bleeding likely from a branch of inferior mesenteric artery (Figs. 1A and B). She underwent emergency embolization of the feeding vessels with gelfoam to stop the active bleeding. Following the embolization, CT showed a lobulated soft tissue density mass in the pelvis measuring about 15 × 6.6 × 6.5 cm which was compressing the rectum and the bladder and with bilateral hydronephrosis (Figs. 2A and B). Serum CA-125 was 595.8 and CA-19.9 was 26.9. Figures 1A and B: Computed tomography (CT) of abdomen, pelvic and chest. A: Large amount of dense free fluid within the pelvis and moderate amount of more hypodense free fluid is also visualized in the abdomen. B: ere is a blush of increasing contrast enhancement in the central and right hemipelvis in keeping with active hemorrhage. Her past surgical history suggested that she had undergone total hysterectomy for fibroid uterus and after two years she had bilateral salpingo-oophorectomy and partial vaginectomy for deeply infiltrating endometriosis. Post-operatively, she was neither treated Oman Medical Journal (2014) Vol. 29, No. 3:226-231 DOI 10.5001/omj.2014.56

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Oman Medical Specialty Board

Endometriosis After Surgical Menopause Mimicking Pelvic Malignancy: Surgeons’ Predicament

Rani A. Bhat, Melissa Teo and Akhil Krishnanand Bhat

Rani A. Bhat Consultant Gynaecological Oncologist HCG Oncology No. 8, P Kalinga Rao Road Sampangi Ramnagar Bangalore - 560027. India.E-mail: [email protected]

Melissa TeoSenior Consultant, Department of Surgical Oncology, National Cancer Centre Singapore, Singapore.

Akhil Krishnanand BhatSenior Consultant-General Surgery, St. Philomena's Hospital, No. 4 Cambell Road, Viveknagar, Bangalore-560047, India.

Abstract

Prevalence of persistent endometriosis in women after menopause without any hormonal replacement therapy is very rare. This is a case of a woman with previous history of total hysterectomy and bilateral salpingo-oophorectomy for endometriosis who presented with hemoperitoneum, vaginal bleeding, pelvic mass, and pulmonary thromboembolism mimicking as rectovaginal septum carcinoma. This is the first case report with a unique mode of presentation wherein the patient presented with hemoperitoneum requiring emergency embolization of the vessel to stabilize the patient. She underwent en bloc resection of the tumor with high anterior resection of the rectum. Histopathology confirmed endometriosis.

Keywords: Endometriosis; Ovarian cancer; Menopause.

Introduction

The incidence of endometriosis in post-menopausal women is 2% to 5%1,2 and is commonly seen in those who have received hormonal replacement therapy after menopause.3 This report is about a rare case of a patient with post-menopausal endometriosis, who presented with intra-abdominal bleeding, pelvic mass, vaginal bleeding and pulmonary thromboembolism. With clinical and radiological findings and past history of endometriosis, the disease was considered as rectovaginal septum carcinoma of clear cell type resulting from malignant transformation of endometriosis.

Received: 10 Feb 2014 / Accepted: 12 Apr 2014© OMSB, 2014

Case Report

A 50-year-old para 2 was referred to our hospital in view of pelvic mass, bilateral hydronephrosis and pulmonary embolism. She had initially presented to a private hospital with a history of acute abdomen due to hemoperitoneum and bleeding per-vagina. A contrast enhanced CT scan of the abdomen and pelvis showed large hemoperitoneum with solid dense areas in the caudal portion of the pelvis with a blush of increasing contrast enhancement in the central part of the pelvis indicating active bleeding likely from a branch of inferior mesenteric artery (Figs. 1A and B). She underwent emergency embolization of the feeding vessels with gelfoam to stop the active bleeding. Following the embolization, CT showed a lobulated soft tissue density mass in the pelvis measuring about 15 × 6.6 × 6.5 cm which was compressing the rectum and the bladder and with bilateral hydronephrosis (Figs. 2A and B). Serum CA-125 was 595.8 and CA-19.9 was 26.9.

Figures 1A and B: Computed tomography (CT) of abdomen, pelvic and chest. A: Large amount of dense free fluid within the pelvis and moderate amount of more hypodense free fluid is also visualized in the abdomen. B: There is a blush of increasing contrast enhancement in the central and right hemipelvis in keeping with active hemorrhage.

Her past surgical history suggested that she had undergone total hysterectomy for fibroid uterus and after two years she had bilateral salpingo-oophorectomy and partial vaginectomy for deeply infiltrating endometriosis. Post-operatively, she was neither treated

Oman Medical Journal (2014) Vol. 29, No. 3:226-231DOI 10.5001/omj.2014.56

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Oman Medical Specialty Board

for her endometriosis nor was she on any hormonal replacement therapy. With this mode of clinical presentation, the differential diagnosis considered were carcinoma of the peritoneum or recto-vaginal septum due to malignant transformation of the remnant endometriosis mainly of clear cell type due to the typical features of pelvic mass, pulmonary embolism and past history of endometriosis.

Figures 2A and B: CT of abdomen, pelvis and chest after embolization of inferior mesenteric artery: Lobulated soft tissue density mass seen in the pelvis measuring about 15 × 6.6 × 6.5 cm, compressing the rectum and bladder and displacing the small bowel loops laterally.

She underwent laparotomy revealing a 5 × 3 × 2.5 cm mass which was densely adherent to the rectosigmoid colon, to the pelvic side walls involving bilateral ureters, and eroding into the vagina causing ulceration of the vaginal vault. There was neither free intraperitoneal fluid nor any signs of pelvic or abdominal endometriosis. Preoperative CT showed a 15 × 6.6 × 6.5 cm mass in the pelvis, but intraoperatively, only a 5 × 3 × 2.5 cm mass was noted in the pelvis giving rise to a possibility that the intraperitoneal bleeding had undergone resorption. She underwent uretrolysis, en bloc resection of the tumor, and high anterior resection of the rectum. Postoperatively, the patient made uneventful recovery and was discharged home on the 9th postoperative day. Final histopathological findings showed features consistent with so-called necrotic pseudoxanthomatous nodule, associated with longstanding endometriosis and no features of malignancy.

Discussion

It has been hypothesized that endometriosis is estrogen-dependent and that progesterone inhibits the cellular proliferation, therefore endometriosis is commonly seen in women of reproductive age group and very rarely seen after menopause. Different theories have been postulated for recurrence of endometriosis in women

who have undergone surgical menopause, such as ovarian remnant syndrome (ORS), wherein part of the ovarian tissue has been left behind after bilateral oophorectomy which continues to produce hormone and stimulate the ectopic endometrial implant. The endometrial implants can also be reactivated by exogenous estrogen in the form of hormonal replacement therapy,3,4 or endogenous estrogen which comes from peripheral conversion of androgen and androstenedione from adrenal glands. Evidence also shows that endometrial implants harbor aromatase expression which is stimulated by PGE2 leading to local production of estrodiol. These implants also lack hydroxysteroid dehydrogenase (17β-HSD) type 2 expression thus impairing conversion of estradiol to estrone which results in local accumulation of potent estrodiol. Another possible hypothesis of endometriosis after menopause is due to spontaneous coelomic metaplasia and vascular endometrial cell transportation. It is possible that our patient had ovarian remnant syndrome because her BMI was normal, had no history of HRT but had past history of deep endometriosis making it possible that part of ovarian tissue must have been left in-situ during laparotomy done for bilateral salpingo-oophorectomy and vaginectomy. Since we did not have preoperative serum estradiol and FSH levels, it was difficult to demonstrate her true menopausal status.

Table 1 shows a summary of studies on endometriosis in women who have undergone surgical menopause. Presentation of endometriosis in post-menopausal patients can be unpredictable and mainly depends on the location of the endometriotic implants. Since endometriosis is usually associated with surrounding inflammation and fibrosis, symptoms can sometimes be very severe. Since the time of Sampson in 1925,5 numerous case reports and review of the literature have shown that endometriosis can be a precursor of ovarian, primary peritoneal or recto-vaginal septum cancer mainly of clear cell, endometroid type.6,7 Literature has also shown that endometriosis can act as a precursor for both cystic and adenofibromatous types of clear cell carcinoma of the ovary.8 Endometriosis can sometimes present with ascites, pelvic mass and pleural effusion mimicking as advanced ovarian cancer,9 thus making preoperative diagnosis of endometriosis, especially in women who have undergone total hysterectomy and bilateral salpingo-oophorectomy, very challenging to the treating physician. Clear cell carcinoma demonstrates unique clinical features such as large pelvic mass, thromboembolic complications and hypercalcemia.

In this case report, the patient had clinical features of pelvic mass, hemoperitoneum and pulmonary embolism and our diagnosis was carcinoma of the recto-vaginal septum of clear cell type associated with endometriosis. Surgical resection of all endometriotic implants and restoring normal anatomy is the treatment of choice. Hence, in patients with deeply infiltrating endometriosis, radical surgery involving bowel resection becomes inevitable. Following surgery postoperative hormonal treatment to suppress endometriosis has not yet been established.10

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Oman Medical Specialty Board

Tab

le 1

: Sum

mar

y of

stud

ies o

f end

omet

rios

is in

wom

en a

fter

tota

l hys

tere

ctom

y w

ith b

ilate

ral s

alpi

ngo-

ooph

orec

tom

y (T

HB

SO).

Cas

e no

Ref

eren

ceA

geM

eno-

paus

e st

atus

Mod

e of

pr

esen

tati

onC

linic

al

feat

ures

Past

his

tory

of

en

dom

etri

osis

Past

his

tory

of

surg

ery

His

tory

of

horm

onal

tr

eatm

ent f

or

endo

met

rios

is

Past

his

tory

of

HR

T**

*P

re-o

pera

tive

diag

nosi

s mad

eT

reat

men

tH

isto

logy

1T

akay

ama

K, Z

eito

un K

, G

unby

RT

, Sas

ano

H, C

arr

BR, B

ulun

SE

. Tre

atm

ent

of se

vere

pos

tmen

opau

sal

endo

met

rios

is w

ith a

n ar

omat

ase

inhi

bito

r.Fe

rtil

Ster

il. 1

998

Apr

;69(

4):7

09-1

3

53Ye

sPe

lvic

pai

nB

ilate

ral

dist

al u

rete

ral

obst

ruct

ion

Yes

TH

BSO

*N

oYe

s (or

al

conj

ugat

ed

estr

ogen

)

End

omet

rios

isE

xcis

ion

of th

e en

dom

etri

osis

and

re

impl

anta

tion

of b

ilate

ral u

rete

r in

to th

e bl

adde

r. R

ecur

renc

e of

en

dom

etri

osis

aft

er 3

ye

ars w

as tr

eate

d w

ith

arom

atas

e in

hibi

tor.

End

omet

rios

is

2G

iare

nis I

, Gia

mou

gian

nis

P, S

peak

man

CT

, Nie

to

JJ, C

rock

er S

G. R

ecur

rent

en

dom

etri

osis

follo

win

g to

tal h

yste

rect

omy

with

oo

phor

ecto

mym

imic

king

a

mal

igna

nt n

eopl

astic

lesi

on:

a di

agno

stic

and

ther

apeu

tic

chal

leng

e.A

rch

Gyn

ecol

Obs

tet.

2009

M

ar;2

79(3

):419

-21.

44Ye

sPa

inle

ss v

agin

al

blee

ding

Pelv

ic m

ass

Yes

TH

BSO

*N

oYe

s (c

onju

gate

d es

trog

en)

Mal

igna

nt

neop

last

ic

lesi

on, p

ossi

bly

a sa

rcom

a

Exc

isio

n of

the

pelv

ic

mas

s with

ant

erio

r re

sect

ion

of th

e si

gmoi

d co

lon.

End

omet

rios

is

3B

aile

y A

P, S

chut

t AK

, M

odes

itt S

C. F

lori

d en

dom

etri

osis

in

post

men

opau

sal w

oman

. Fer

til

Ster

il. 2

010

Dec

;94(

7):2

769.

e1-4

. Epu

b 20

10 M

ay 2

6.

53Ye

sG

ross

he

mat

uria

Rig

ht si

ded

retr

oper

itone

al

mas

s cau

sing

se

ver h

ydro

-ur

eter

o-ne

phro

sis

Yes

TH

BSO

*N

oN

oN

ot m

entio

ned

Rig

ht si

mpl

e ne

phre

ctom

y, ra

dica

lre

sect

ion

of th

e re

trop

erito

neal

mas

s in

clud

ing

diss

ectio

n of

par

tof

the

psoa

s mus

cle

and

the

infe

rior

ven

a ca

va, r

esec

tion

of th

e di

stal

ileum

plu

s cec

um

and

appe

ndix

due

to

mes

ente

ric

inva

sion

, an

d pr

imar

yile

oasc

endi

ng c

olon

re

anas

tom

osis

End

omet

rios

is

Oman Medical Journal (2014) Vol. 29, No. 3:226-231

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Oman Medical Specialty Board

Oman Medical Journal (2014) Vol. 29, No. 3:226-231

Cas

e no

Ref

eren

ceA

geM

eno-

paus

e st

atus

Mod

e of

pr

esen

tati

onC

linic

al

feat

ures

Past

his

tory

of

en

dom

etri

osis

Past

his

tory

of

surg

ery

His

tory

of

horm

onal

tr

eatm

ent f

or

endo

met

rios

is

Past

his

tory

of

HR

T**

*P

re-o

pera

tive

diag

nosi

s mad

eT

reat

men

tH

isto

logy

4In

drac

colo

U, B

arbi

eri F

. Sile

nt

onse

t of p

ostm

enop

ausa

l en

dom

etri

osis

ina

wom

an w

ith re

nal f

ailu

re in

ho

rmon

e re

plac

emen

t the

rapy

: a

case

repo

rt. J

Med

Cas

e R

epor

ts. 2

010

Aug

4;4

:248

.

54Ye

sR

enal

failu

reB

ilate

ral

hydr

onep

hros

is

indu

ced

byex

trin

sic

com

pres

sion

of

both

ure

ters

(at

supr

aves

ical

foss

a) b

y no

dule

s co

mpa

tible

Yes

TH

BSO

*N

oYe

s (es

trog

en

base

d on

ly)f

or

seve

n ye

ars

End

omet

rios

isla

paro

scop

ic e

xcis

ion

of th

e en

dom

etri

otic

no

dule

s

End

omet

rios

is

5K

hong

SY,

Lam

A, C

oom

bes

G, F

ord

S. S

urgi

cal

man

agem

ent o

f rec

urre

nt

uret

eric

end

omet

rios

is c

ausi

ng

recu

rren

t hyp

erte

nsio

n in

a

post

men

opau

sal w

oman

.J M

inim

Inva

sive

Gyn

ecol

. 20

10 Ja

n-Fe

b;17

(1):1

00-3

.

62Ye

sR

ecur

rent

ur

inar

y tr

act

infe

ctio

n an

d pa

in in

left

ilia

c fo

ssa

Obs

truc

ted

left

ure

ter a

nd

hydr

onep

hros

is

Yes

TH

BSO

*N

oYe

s (es

trog

en

patc

h)E

ndom

etri

osis

Exc

isio

n of

en

dom

etri

otic

nod

ule

End

omet

rios

is

6Po

pout

chi P

, dos

Rei

s Lem

os

CR

, Silv

a JC

, Nog

ueir

a A

A,

Fere

s O, R

ibei

ro d

aR

ocha

JJ: P

ostm

enop

ausa

l in

test

inal

obs

truc

tive

endo

met

rios

is: c

ase

repo

rt a

nd re

view

of t

he

liter

atur

e. Sa

o Pa

olo

Med

J 20

08, 1

26:1

90-1

93.

74Ye

sH

emat

oche

zia,

te

nesm

us a

nd

pelv

ic p

ain

Col

onos

copy

re

veal

ed a

fr

iabl

e an

d st

enos

ing

tum

or

form

atio

n in

the

uppe

r rec

tum

.

Yes

TH

BSO

*N

oN

oA

bio

psy

reve

aled

m

ucos

al

frag

men

ts o

f en

dom

etri

al

type

Abd

omin

al

rect

osig

moi

dect

omy

with

a lo

w m

echa

nica

l co

lore

ctal

ana

stom

os

and

tran

sver

sost

omy

in a

pro

tect

ive

loop

pe

rfor

med

.

End

omet

rios

is

7R

. Fly

ckt,

S. L

yden

, A. R

oma

and

T. F

alco

ne*

Post

-men

opau

sal

endo

met

rios

is w

ith in

feri

or

vena

cav

a in

vasi

on re

quir

ing

surg

ical

man

agem

ent

Hum

Rep

rod.

201

1, 2

6 (1

0):2

709-

2712

.doi

: 10.

1093

/hu

mre

p/de

r260

59Ye

sLe

ft lo

wer

ab

dom

inal

pai

nPe

ri-a

ortic

mas

s w

ith u

rete

ral

obst

ruct

ion

YE

ST

HB

SO*

No

Yes

(Con

juga

ted

equi

ne

estr

ogen

)

CT

** g

uide

d bi

opsy

reve

aled

en

dom

etri

osis

Exc

isio

n of

the

tum

or

with

rese

ctio

n an

d lig

atio

n of

infe

rior

ve

na c

ava

End

omet

rios

is

Tab

le 1

: Sum

mar

y of

stud

ies o

f end

omet

rios

is in

wom

en a

fter

tota

l hys

tere

ctom

y w

ith b

ilate

ral s

alpi

ngo-

ooph

orec

tom

y (T

HB

SO).

-cont

inue

d

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230

Oman Medical Specialty Board

Cas

e no

Ref

eren

ceA

geM

eno-

paus

e st

atus

Mod

e of

pr

esen

tati

onC

linic

al

feat

ures

Past

his

tory

of

en

dom

etri

osis

Past

his

tory

of

surg

ery

His

tory

of

horm

onal

tr

eatm

ent f

or

endo

met

rios

is

Past

his

tory

of

HR

T**

*P

re-o

pera

tive

diag

nosi

s mad

eT

reat

men

tH

isto

logy

8Jo

seph

J, R

eed

CE

, Sa

hn S

A. Th

orac

ic

endo

met

rios

is. R

ecur

renc

e fo

llow

ing

hyst

erec

tom

y w

ith b

ilate

rals

alpi

ngo-

ooph

orec

tom

y an

d su

cces

sful

tr

eatm

ent w

ith ta

lc

pleu

rode

sis.

Che

st. 1

994

Dec

;106

(6):1

894-

6.

30Ye

sIn

term

itten

tri

ght-

and

left

-si

ded

pleu

ritic

ch

est p

ain,

he

mop

tysi

s of

6-ye

ars'

dura

tion,

and

re

cent

ple

ural

eff

usio

n.

Dec

reas

ed v

ocal

fr

emitu

s and

di

min

ishe

d br

eath

soun

ds in

th

e le

ft b

ase,

Thor

acen

tesi

s re

veal

eda

hem

orrh

agic

flu

id

Yes

TH

BSO

* +

pa

st h

isto

ry o

f th

orac

otom

y an

d ex

cisi

on o

f ri

ght l

ung

bleb

san

d pl

eura

l ab

rasi

on

wer

e do

ne

for r

ecur

rent

pn

eum

otho

rax

for r

ecur

rent

en

dom

etri

osis

.

Yes (

dana

zol)

Yes (

estr

ogen

an

d pr

oges

tero

ne)

End

omet

rios

isT

alc

Ple

urod

esis

for

recu

rren

t tho

raci

c en

dom

etri

osis

End

omet

rios

is

9R

ana

N, R

otm

an C

, H

asso

n H

M, R

edw

ine

DB

, D

mow

ski W

P. O

vari

an

rem

nant

synd

rom

e af

ter

lapa

rosc

opic

hys

tere

ctom

y an

d bi

late

rals

alpi

ngo-

ooph

orec

tom

y fo

r sev

ere

pelv

ic

endo

met

rios

is.

J Am

Ass

oc G

ynec

ol L

apar

osc.

1996

May

;3(3

):423

-6.

33Ye

sV

agin

al

blee

ding

and

pe

lvic

pai

n

Cys

tic le

sion

in

the

pelv

isYe

sH

ad

Supr

acer

vica

l hy

ster

ecto

my

and

BSO

fo

llow

ing

faile

d m

edic

al

man

agem

ent

for

endo

met

rios

is,

but h

ad

recu

rren

ce in

sp

ite o

f med

ical

m

anag

emen

t of

endo

met

rios

is

Yes

(cyc

lic-

nore

thin

o-dr

one

acet

ate)

Yes (

ethi

nyl

estr

adio

l)E

ndom

etri

osis

Vag

inal

rese

ctio

n of

th

ece

rvic

al st

ump

and

lapa

rosc

opic

rese

ctio

n of

the

ovar

ian

rem

nant

.

End

omet

rios

is

*TH

BSO

: Tot

al h

yste

recto

my

with

bila

tera

l sal

ping

o-oo

phor

ecto

my,

**C

T: C

ompu

ted to

mog

raph

y, **

*HRT

: Hor

mon

al re

plac

emen

t the

rapy

Tab

le 1

: Sum

mar

y of

stud

ies o

f end

omet

rios

is in

wom

en a

fter

tota

l hys

tere

ctom

y w

ith b

ilate

ral s

alpi

ngo-

ooph

orec

tom

y (T

HB

SO).

-cont

inue

d

Oman Medical Journal (2014) Vol. 29, No. 3:226-231

Page 6: Endometriosis After Surgical Menopause … › imemrf › Oman_Med_J › Oman...Endometriosis After Surgical Menopause Mimicking Pelvic Malignancy: Surgeons’ Predicament Rani A

231

Oman Medical Specialty Board

Oman Medical Journal (2014) Vol. 29, No. 3:226-231

Conclusion

Endometriosis after surgical menopause is rare and symptoms can vary based on the site of endometriotic implants. Sometimes endometriosis can present with symptoms which can mimic pelvic malignancies and also since endometriosis confers the risk of malignant transformation, this makes it difficult for a treating physician to make appropriate pre-operative diagnosis. Although endometriosis after surgical menopausal is rare, it should be considered in the differential diagnosis of abdominal or pelvic mass, especially in women with past history of endometriosis.

Acknowledgements

We would like to thank Professor Soo Khee Chee and A / Prof Koong Heng Nung for their help with the design of the study protocol. No conflict of interests or funding to declare.

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