identifying and assessing the benefits of interventions ... · health care systems. the comparators...

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RESEARCH ARTICLE Open Access Identifying and assessing the benefits of interventions for postnatal depression: a systematic review of economic evaluations Binu Gurung 1 , Louise J. Jackson 1* , Mark Monahan 1 , Ruth Butterworth 2 and Tracy E. Roberts 1 Abstract Background: Economic evaluations of interventions for postnatal depression (PND) are essential to ensure optimal healthcare decision-making. Due to the wide-ranging effects of PND on the mother, baby and whole family, there is a need to include outcomes for all those affected and to include health and non-health outcomes for accurate estimates of cost-effectiveness. This study aimed to identify interventions to prevent or treat PND for which an economic evaluation had been conducted and to evaluate the health and non-health outcomes included. Methods: A systematic review was conducted applying a comprehensive search strategy across eight electronic databases and other sources. Full or partial economic evaluations of interventions involving preventive strategies (including screening), and any treatments for women with or at-risk of PND, conducted in OECD countries were included. We excluded epidemiological studies and those focussing on costs only. The included studies underwent a quality appraisal to inform the analysis. Results: Seventeen economic evaluations met the inclusion criteria, the majority focused on psychological /psychosocial interventions. The interventions ranged from additional support from health professionals, peer support, to combined screening and treatment strategies. Maternal health outcomes were measured in all studies; however child health outcomes were included in only four of them. Across studies, the maternal health outcomes included were quality-adjusted-life-years gained, improvement in depressive symptoms, PND cases detected or recovered, whereas the child health outcomes included were cognitive functioning, depression, sleep and temperament. Non-health outcomes such as couplesrelationships and parent-infant interaction were rarely included. Other methodological issues such as limitations in the time horizon and perspective(s) adopted were identified, that were likely to result in imprecise estimates of benefits. Conclusions: The exclusion of relevant health and non-health outcomes may mean that only a partial assessment of cost-effectiveness is undertaken, leading to sub-optimal resource allocation decisions. Future research should seek ways to expand the evaluative space of economic evaluations and explore approaches to integrate health and non-health outcomes for all individuals affected by this condition. There is a need to ensure that the time horizon adopted in studies is appropriate to allow true estimation of the long-term benefits and costs of PND interventions. Keywords: Postpartum depression, Economic analysis, Parental outcomes, Non-health consequences, QALYs, Outcomes for young children * Correspondence: [email protected] 1 Health Economics Unit, Institute of Applied Health Research, College of Medical and Dental Sciences, University of Birmingham, Edgbaston, Birmingham B15 2TT, UK Full list of author information is available at the end of the article © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the 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. Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 https://doi.org/10.1186/s12884-018-1738-9

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Page 1: Identifying and assessing the benefits of interventions ... · health care systems. The comparators included placebo, no intervention and current or standard care. In terms of study

RESEARCH ARTICLE Open Access

Identifying and assessing the benefits ofinterventions for postnatal depression: asystematic review of economic evaluationsBinu Gurung1, Louise J. Jackson1*, Mark Monahan1, Ruth Butterworth2 and Tracy E. Roberts1

Abstract

Background: Economic evaluations of interventions for postnatal depression (PND) are essential to ensure optimalhealthcare decision-making. Due to the wide-ranging effects of PND on the mother, baby and whole family, thereis a need to include outcomes for all those affected and to include health and non-health outcomes for accurateestimates of cost-effectiveness. This study aimed to identify interventions to prevent or treat PND for which aneconomic evaluation had been conducted and to evaluate the health and non-health outcomes included.

Methods: A systematic review was conducted applying a comprehensive search strategy across eight electronicdatabases and other sources. Full or partial economic evaluations of interventions involving preventive strategies(including screening), and any treatments for women with or at-risk of PND, conducted in OECD countrieswere included. We excluded epidemiological studies and those focussing on costs only. The included studiesunderwent a quality appraisal to inform the analysis.

Results: Seventeen economic evaluations met the inclusion criteria, the majority focused on psychological /psychosocialinterventions. The interventions ranged from additional support from health professionals, peer support, to combinedscreening and treatment strategies. Maternal health outcomes were measured in all studies; however child healthoutcomes were included in only four of them. Across studies, the maternal health outcomes included werequality-adjusted-life-years gained, improvement in depressive symptoms, PND cases detected or recovered,whereas the child health outcomes included were cognitive functioning, depression, sleep and temperament.Non-health outcomes such as couples’ relationships and parent-infant interaction were rarely included. Othermethodological issues such as limitations in the time horizon and perspective(s) adopted were identified, thatwere likely to result in imprecise estimates of benefits.

Conclusions: The exclusion of relevant health and non-health outcomes may mean that only a partial assessment ofcost-effectiveness is undertaken, leading to sub-optimal resource allocation decisions. Future research should seek waysto expand the evaluative space of economic evaluations and explore approaches to integrate health and non-healthoutcomes for all individuals affected by this condition. There is a need to ensure that the time horizon adopted instudies is appropriate to allow true estimation of the long-term benefits and costs of PND interventions.

Keywords: Postpartum depression, Economic analysis, Parental outcomes, Non-health consequences, QALYs,Outcomes for young children

* Correspondence: [email protected] Economics Unit, Institute of Applied Health Research, College ofMedical and Dental Sciences, University of Birmingham, Edgbaston,Birmingham B15 2TT, UKFull list of author information is available at the end of the article

© The Author(s). 2018 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.

Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 https://doi.org/10.1186/s12884-018-1738-9

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BackgroundPostnatal depression (PND), also called postpartum de-pression, is a non-psychotic, depressive disorder occur-ring in women within a year after childbirth [1, 2]. It is acommon disorder thought to affect 1 in 10 womenwithin the first postpartum year [2]. The period preva-lence of minor or major depressive episodes is estimatedto be 19.2% in the first three months following child-birth, with 7.1% of mothers experiencing major depres-sive episodes [3]. Mothers with PND are likely toexperience disturbing emotions and feelings common todepression such as anger, guilt, hopelessness, social with-drawal, and those specific to the perinatal period such assleep deprivation and bonding difficulty [4, 5].As well as affecting the mother, PND can also affect

others within the family. A meta-analysis of studies doc-umenting depression of fathers in the first postpartumyear reported that fathers had a 10% risk of experiencingdepression and found the correlation between paternaland maternal depression to be positive and moderate[6]. The experience of PND in the parent or parents canpotentially lead to marital problems, the withdrawal ofsocial support between parents can compromise ad-equate care-giving practices of parents, or parent-babyinteractions (e.g. the ability of the mother to respondsensitively to her child), that in turn may negativelyaffect the cognitive, behavioural and social developmentof the infant in the short and long-term [7–9].The economic costs associated with PND are signifi-

cant [10]. In 2002, Petrou and colleagues reported aver-age additional health and social care costs of £392 (2000prices; UK pound sterling) in women with PND com-pared to women without PND over the first 18 monthspost-partum [11]. Recently, Bauer and colleagues esti-mated that the societal discounted cost of depressionduring pregnancy and the postnatal period in the long-term was nearly £74,000 per case (2012/13 prices; UKpound sterling) [12]. Around 70% of the projected costsrelated to the impacts on children, calculated in terms ofpre-term birth, mortality, emotional problems, educationand conduct, over a period ranging between birth andoverall lifetime.PND has long been considered a major public health

problem [13, 14] and a range of PND interventions havebeen developed in order to prevent or treat the condi-tion. Compared to evaluations of clinical effectiveness ofPND interventions, evaluation of their cost-effectivenesswithin an economic evaluation (comparative analysis ofalternative interventions or programmes in terms ofboth costs and consequences) has been relatively limited[15–17]. The economic evaluation of a PND interventionis essential to understand the value of the interventionrelative to other interventions to allow appropriate alloca-tion of healthcare resources [18]. A key consideration in

an economic evaluation of an intervention in health con-ditions like PND, where the impact could potentially gobeyond mothers to children, fathers, and could includenon-health aspects such as the child’s educational andemotional well-being, is ensuring that all relevant out-comes for all those affected by the intervention are identi-fied and included [19]. Furthermore, it may be necessaryto include outcomes that are broader than typical directhealth outcomes, as is often the case for public healthinterventions [20, 21].Therefore this study was conducted with the aim to

systematically review published and unpublished studiesof interventions to treat or prevent PND, in which aneconomic evaluation has been conducted in OECD(Organisation for Economic Cooperation and Develop-ment) countries, in order to investigate the outcomesconsidered and measured. The specific objectives were i)to identify studies of interventions to prevent or treatPND which included an economic evaluation; ii) to as-certain which outcomes were included and how thesewere measured and valued; and iii) to identify anymethodological issues associated with including andmeasuring outcomes in economic evaluations of PNDinterventions.

MethodsA systematic review was conducted in adherence withguidance on methods from the Centre for Review andDissemination [22] and on reporting from PreferredReporting of Items for Systematic Review and Meta-Analysis (PRISMA) [23].

Search strategyA comprehensive, systematic search strategy was de-veloped through consultation with an informationspecialist (Additional file 1). The searches were runfrom database inception to July 2015 in eight health-care databases: MEDLINE, MEDLINE in-process andother non-indexed citations, EMBASE, PsychINFO,Cumulative Index to Nursing & Allied Health Litera-ture (CINAHL), National Health Service Economicevaluation database (NHS EED), Health TechnologyAssessment (HTA) and Web of Science (WOS) corecollections. The search was not restricted by thepublication date or language. Alongside this process,key journals were hand-searched (these were thosewhich appeared most frequently in results of thesearches for relevant papers) and reference lists ofall the included studies were screened. Furthermore,key researches in the field and members of theBirmingham Perinatal and Infant Mental HealthForum were contacted to identify potential publishedor unpublished literature.

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Inclusion and exclusion criteriaThe studies were assessed and selected using the PICOSframework [22] as a guide. The inclusion criteria were:women with or ‘at risk’ of postnatal depression (i.e. thosewho are pregnant or have given birth within the12 months), living in OECD countries, and interventionsinvolving preventive strategies (including screening), andany treatments or other interventions for PND. Werestricted our focus to OECD countries, in order tocompare economic evaluations concerned with similarhealth care systems. The comparators included placebo,no intervention and current or standard care. In termsof study design, studies that involved a full or partialeconomic evaluation or that included economic datawere potentially eligible for inclusion.Broadly speaking, the different forms of economic

evaluation can be differentiated by how outcomes areconsidered (although there are also other key differ-ences, for example, in terms of their theoretical founda-tions) [10]. A cost-utility analysis (CUA) involvesconsideration of outcomes in terms of quality-adjusted-life-years (QALYs) which combine measurement ofquantity and quality of life [18]. In cost-effectivenessanalysis (CEA) outcomes are expressed in natural units(e.g. cases detected) and in cost-benefit analysis (CBA)outcomes are valued in monetary terms. Partial forms ofeconomic evaluation include cost-consequence analysis(CCA) where costs and outcomes are presented in adisaggregated form and cost-minimisation analysis(CMA) that is only recommended in certain circum-stances where treatments are proven to have identicaloutcomes [10].The following were excluded from the review: epi-

demiological studies reporting incidence/prevalence;costing studies describing costs only; clinical studiesdescribing and evaluating efficacy or effectiveness only;ongoing or incomplete economic evaluations; discussionpapers, letters or commentaries.

Study categorisationA two-stage process outlined by Roberts and colleagues[24] was used to select and categorise studies based ontheir eligibility and codes for Stage 1 and 2 are providedin Additional file 2 (and as a Footnote for Fig. 1). Stage1 involved the initial categorisation of studies into cat-egories A to E based on titles and abstracts according towhether the study involved an economic evaluation orincluded economic data. One author (BG) carried outthe initial coding of the studies and another author (LJ)checked the coding. Studies categorised as potentiallyrelevant to the systematic review were carried forwardto Stage 2 and assessed for inclusion based on theirfull-text. One author (BG) carried out the stage 2coding from 1 to 7, which was then checked by two

authors (LJ and MM) and two authors (BG, LJ)assessed the full-texts of all the studies carried for-ward to Stage 2.

Data extraction and quality assessmentA data extraction form was used to collect data on thebackground of the study, details of the outcomes in-cluded and any methodological limitations acknowl-edged by study authors (Additional file 3). The includedstudies were assessed for quality using an adapted ver-sion of Drummond et al.’s checklist [25], which is judgedto be suitable for assessing economic evaluations [22].According to the objectives of the review, the modifica-tions to the checklist included greater focus on outcomesthan costs, the addition of a question on the perspectiveof analysis and the removal of a general question on thestudy results. The assessment aimed to inform the mainanalysis rather than to exclude studies based on quality(the full results are available in Additional file 4).

ResultsThe electronic search of the databases yielded 2360 stud-ies. A further eight studies were identified as potentiallyrelevant to the review from other sources (key researchersin this field and the forum members). After removing 537duplicates, 1831 records were categorised based on titleand abstract (Stage 1). The full-texts of 58 papers wereassessed (Stage 2) and 17 studies were identified forsynthesis. Figure 1 presents a PRISMA flow diagram ofthe articles screened, included or excluded in each stage.

Study characteristicsThe study characteristics are provided in Table 1. Thestudies were predominantly from the UK (n = 13) with afurther four studies conducted in Australia [26], Canada[27], New Zealand [28] and the USA [29].A variety of interventions were evaluated in the identi-

fied studies that conducted an economic evaluation. Ninestudies examined interventions concerned with preventivestrategies only [26, 27, 29–35], five studies included bothscreening and treatment [28, 30, 36–38], and theremaining three studies assessed treatment only [39–41].The most common type of intervention was modified orenhanced support or care in the perinatal period eitherfrom a health professional or via peer support [27, 28, 30–35, 41]. Many of the interventions included cognitivebehavioural therapy (CBT), either alone [40], or incombination with other therapies such as antidepressants[42], interpersonal therapy [29], listening visits [37, 38]and a range of customised treatments [39]. A pharmaco-logical intervention was delivered as one of the interven-tions in three studies only [35, 39, 42]. Various screeningtools (Edinburgh Postnatal Depression Scale - EPDS,Whooley questions, Beck Depression Inventory, PHQ-9)

Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 3 of 18

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were assessed as part of a wider strategy to identify andtreat those experiencing PND [28, 36, 38].The comparatoradopted was usual, standard or current care/practice/ser-vices in nearly all studies. Two studies used ‘no interven-tion’ as the comparator [30, 34]. Seven studies did notprovide a comprehensive description of their comparators[26, 31, 32, 35, 40–42].All studies investigating screening considered women

in their postnatal period [28, 36–38, 42]. Other prevent-ive strategies focused on pregnant [31, 33] and/orpostpartum women [26–28, 30, 33, 35]. For treatmentinterventions, the targeted population were postpartum

women with a PND diagnosis and/or women scoringabove the threshold of a screening tool [28, 36, 38–42].There were some differences in terms of the aims of

the interventions evaluated. All but three studies evalu-ated an intervention that focussed on preventing and/ortreating PND alone. These three studies focused onimproving other aspects of health and well-being inaddition to addressing PND, including women’s physicaland general health [31, 32], maternal smoking [35] andchild injury [35].There was a greater number of studies that conducted an

economic evaluation alongside a Randomised Controlled

Fig. 1 PRISMA flowchart showing the study selection process.Notes: Coding- Stage 1) A. The study involves a formal economic evaluation of PND interventions based on primary and/or secondary data (e.g.previously published studies or other sources); B. The study discusses economic aspects of PND interventions and contains relevant primary and/or secondary data; C. Unclear if the study falls under (A) or (B) but contains useful information; D. The study discusses economic aspects of PNDinterventions, but is neither (A) nor (B); E. The study is not relevant to the economic evaluation of PND interventions. Stage 2) 1. Full economicevaluation; 2. Partial economic evaluation; 3. Study that measured/valued outcomes of PND interventions but did not consider cost or cost-effectiveness; 4. Other, such as study estimating resource use and/or economic burden of PND and interventions; 5. Secondary study discussingmethods or results of economic evaluation; 6. Incomplete economic evaluation of PND interventions (e.g. ongoing studies); 7. Not relevant to theeconomic evaluation of PND interventions. (See Additional file 2)

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Table

1Stud

ycharacteristicsandaims

Lead

author

(Year)

Interven

tion

Com

parator

Cou

ntry

Samplesize,N

;patient

popu

latio

nPrim

aryaimsof

econ

omic

evaluatio

nAnalytical

approach

RCTor

coho

rt-based

econ

omicevaluatio

n

Boath(2003)

[39]

PBDUcustom

ised

treatm

ent(con

sisting

ofon

eor

moreof

thefollowing:

individu

al,

coup

leandfamily

coun

selling

,group

therapy,

creativetherapy,ho

bbiesandactivities,stress

managem

ent,assertiven

esstraining

,yog

aand

relaxatio

n,agrou

pforparentsandolde

rchildren

andph

armacothe

rapy)

RPC

UK

N=60;W

omen

with

ababy

aged

6weeks-1

year,

with

theEPDSscoreabove12

andwith

adiagno

sisof

amajor

orminor

dep

ressivedisorder.

Assessthecost-effectiven

ess

oftw

oalternativeapproaches

tothePN

Dtreatm

ent.

CEA

a

Dukho

vny(2013)

[27]

Teleph

one-basedpe

ersupp

ortinterven

tion,

access

tostandard

postpartum

care

Usualcare

Canada

N=610;High-riskwom

enwith

EPDSscore>9,

ableto

speakEnglish

,followingalivebirthand

dischargeho

me

Determinethecost-effectiven

essof

ape

ersupp

ortinterven

tionto

preven

tPN

D.

CEA

Hiscock

(2007)

[26]

Individu

alstructured

maternaland

child

health

consultatio

ns,a

choice

ofbe

haviou

ralinterventions,

‘con

trolledcrying

’or‘cam

ping

out’

Usualcare

Australia

N=328;Mothe

rsrepo

rtingan

infant

sleepprob

lem

intheconcurrent

7-mon

thqu

estio

nnaire

Assesstheeffectiven

essandcosts

ofan

interven

tiontargetinginfant

sleepprob

lems.

CCA

MacArthu

r(2003)

[31]

Rede

sign

edmod

elof

commun

itypo

stnatalcare

(midwifery-led)

Current

care

UK

N=36

clusters;A

llpreg

nant

wom

enregistered

atthepracticeantenatally

from

abou

t34

weeks

gestation.

Develop

,implem

entandtestthecost-

effectiven

essof

rede

sign

edpo

stnatalcare

comparedwith

curren

tcare

onwom

en’s

physicalandpsycho

logicalh

ealth

.

CCA

Morrell(2000)

[32]

Com

mun

itymidwifery

supp

ortworker

Postnatal

midwifery

care

UK

N=471;Allwom

enwho

delivered

ababy

atthe

recruitin

gho

spitallivingwith

inthestud

yarea,

wereaged

17or

over,and

couldun

derstand

English.

Measure

theeffect

andthetotalcostpe

rwom

anof

providingpo

stnatalsup

port

atho

me.

CMA

Morrell(2009)

[37]

Health

visitortraine

dto

iden

tifyandde

liver

CBA

orpe

rson

-cen

tred

approach

Health

visitor

usualcare

UK

N=418;Wom

enat

riskof

PNDindicatedby

EPDS

score≥12,reg

isteredwith

participatingGPpractices,

36weeks

preg

nant

durin

grecruitm

ent,who

hada

livebaby

andwereon

acollabo

ratin

gHealth

Visitor’s

caseload.

Investigateou

tcom

esforpo

stnatalwom

enattributed

totheinterven

tion,an

dto

establishits

cost-effe

ctiven

ess.

CUAa

Petrou

(2006)

[33]

Cou

nsellingandsupp

ortpackageby

traine

dhe

alth

visitors

RPC

UK

N=151;Wom

enattend

ingclinicsat

26–28weeks

ofge

station,

iden

tifiedas

athigh

riskof

developing

PND

Assessthecost-effe

ctiven

essof

apreventivecoun

selling

andsuppo

rtpackageforwom

enat

highriskof

develop

ingPN

D.

CEA

Price(2015)

[29]

Enhanced

engage

men

tin

homevisitin

gvia

motivationalinterview

ingandbriefinterven

tion

(CBT

andInterpersonalThe

rapy)

Usualcare

USA

N=25;Pregn

antandpo

stpartum

wom

enin

low-

incomeandethn

icminorities

commun

ities

meetin

griskcriteria

formajor

depression

Exam

inethefeasibility

ofen

hanced

engage

men

tin

routinecommun

itycare

over

usualcarematernaland

child

health

homevisitin

g.

CCAa

Sembi

(2016)

[41]

Teleph

onepe

ersupp

ort

Standard

care

UK

N=28;W

omen

>16

yearswhe

ngiving

birth,

expe

riencingde

pressive

symptom

indicatedby

EPDSthreshold≥10

and/or

clinicaljudg

emen

t,andpo

tentially

receptiveto

receivinginterven

tion.

Pilotateleph

onepe

er-sup

portinterven

tion

forwom

enexpe

riencingPN

D.

CCAa

Wiggins

(2004)

[35]

Health

visitorsupp

ortor

Com

mun

itygrou

psupp

ort

Standard

services

UK

N=498;Wom

enlivingin

deprived

areas,who

gave

birthin

thespecified

timeperiodandof

anyethn

icity.

Measure

theim

pact

andcost-effectiven

ess

oftw

oalternativestrategies

forproviding

supp

ortto

mothersindisadvantagedinner

cityarea.

CCAa

Mod

el-based

econ

omicevaluatio

n

Battye

(2012)

[30]

Befriending

service(te

leph

onehelplineand

one-to-one

supp

ortby

trained‘befriend

er’

volunteers)

No

interven

tion

UK

Quantitativestud

y:N=39

Qualitativestud

y:Nno

tprovided

Dem

onstrate

valueformon

eyof

Acacia

Family

Supp

ort’s

service.

CBA

Bauer(2011)

[42]

Universalhe

alth

visitin

g(postnatalscreen

ing

usingEPDSandtreatm

ent[CBT

+antid

epressant])

Routine

postnatalcare

UK

Iden

tification:

Hypothe

ticalcoho

rtof

wom

enforscreen

ing.

Iden

tifyandanalysethecostsandecon

omic

pay-offsof

PNDinterven

tions.

CUAa

Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 5 of 18

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Table

1Stud

ycharacteristicsandaims(Con

tinued)

Lead

author

(Year)

Interven

tion

Com

parator

Cou

ntry

Samplesize,N

;patient

popu

latio

nPrim

aryaimsof

econ

omic

evaluatio

nAnalytical

approach

Treatm

ent:

Hypothe

ticalcoho

rtof

wom

enwith

mod

erate

tosevere

PNDforthetreatm

ent

Cam

pbell(2008)[28]

Routinescreen

ing

prog

ramme(using

PHQ-2)andtreatments

(antidepressants,psychologicaltherapies

orsocialsupport)accordingto

severityofPN

D.

Current

practice

New

Zealand

Nno

tprovided

;Mothe

rswho

gave

birthin

any12

mon

thpe

riod,

regardless

ofthe

numbe

rof

previous

births

Evaluate

valueformon

eyof

implem

entin

gascreen

ingprog

rammeforPN

D.

CUAa ,

CEA

a

Hew

itt(2009)

[36]

Iden

tification

1.EPDS

2.Beck

Dep

ressionInventory

Treatm

ents

1.Structured

psycho

logicalthe

rapy

2.Listen

ingvisit

(Bothwith

preced

ingadditio

nalcare)

Current

practice

UK

Iden

tification:

Hypothe

ticalpo

pulatio

nof

postnatalw

omen

(dep

ressed

orno

t)managed

inprim

arycare

sixweeks

postnatally

Treatm

ent:

Hypothe

ticalcoho

rtsof

1000

wom

enwith

depression

inthepo

stnatalp

eriod

Iden

tification:

Evaluate

thecost-effectiven

essforarang

eof

feasibleiden

tificationstrategies

forPN

Din

prim

arycare.

Treatm

ent:

Clarifyfro

mtheNICEgu

idance

whe

ther

treatm

entstrategies

werecost-effective

comparedwith

usualcare.

CUA

NCCMH(2014)

[38]

Iden

tification

1.EPDSon

ly2.Who

oley

questio

nsfollowed

byEPDS

3.Who

oley

questio

nsfollowed

byPH

Q-9

Treatm

ent

1.Facilitated

self-he

lpbasedon

CBT

principles

2.Listen

ingvisits

(Bothin

additio

nto

standard

postnatalcare)

Standard

care

UK

Iden

tification:

Hypothe

ticalcoho

rtsof

1000

postnatalw

omen

unde

rgoing

screen

ingforde

pression

.Treatm

ent:

Hypothe

ticalcoho

rtsof

1000

wom

enwith

sub-threshold/mild

tomod

eratedepressio

n

Iden

tification:

Assesstherelativecost-effectiven

essof

form

aliden

tificationmetho

dsforPN

D.

Treatm

ent:

Assessthecost-effectiven

essof

different

type

sof

psycho

logicaland

psycho

social,

relativeto

standard

postnatalcarealon

e.

CUAa ,

CEA

a

Steven

son(2010)

[40]

Group

CBT

RPC

UK

Second

aryRC

TN=45;W

omen

meetin

ga

standardised

PNDdiagno

sisor

scoringEPDS

threshold≥12.

Evaluate

theclinicaleffectiven

essand

cost-effectiven

essof

grou

pCBT

compared

with

curren

tlyused

packages.

CUAa

Taylor

(2014)

[34]

Socialsupp

ort(e.g.advocacy,be

frien

ding

)No

interven

tion

UK

Estim

ated

N=100;Wom

enassessed

asvulnerable

toPN

D,eith

eras

aself-referral,orreferredviathe

mid-wife/GP.

Determinethebe

nefitsandcostsof

the

Perin

atalSupp

ortProjectto

preven

tPN

D.

CBA

a

CBACost-Be

nefit

Ana

lysis,CB

ACog

nitiv

eBe

haviou

ralA

pproach,

CBTCog

nitiv

eBe

haviou

ralT

herapy

,CCA

Cost-Con

sequ

ence

Ana

lysis,CE

ACost-Effectiven

essAna

lysis,CM

ACost-Minim

isationAna

lysis,CU

ACost-Utility

Ana

lysis,EPDSEd

inbu

rghPo

stna

talD

epressionScale,

NCC

MHNationa

lCollabo

ratin

gCen

treforMen

talH

ealth

,PBD

UPa

rent

baby

dayun

it,PH

QPa

tient

Health

Que

stionn

aire,R

CTRa

ndom

ised

Con

trolledTrial,RP

CRo

u-tin

eprim

arycare

a Not

explicitlystated

byau

thors

Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 6 of 18

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Trial (RCT) [26, 27, 31–33, 35, 37, 41] or a cohort study[29, 39] compared to studies that undertook decision mod-elling [28, 30, 34, 36, 38, 40, 42]. Different approaches toanalysis were adopted. CUA was the main approachadopted by six of the 17 studies [28, 36–38, 40, 42]. Ofthese six studies, two further conducted a CEA [28, 38].Five of the 17 studies adopted a CCA [26, 29, 31, 35, 41]and three studies conducted a CEA only [27, 33, 39].Two studies carried out a CBA [30, 34] and only onestudy conducted a CMA [32].

Health and non-health outcomesDifferent types of both health and non-health out-comes were included in the economic evaluations(Tables 2 and 3). All economic evaluations includedmaternal health outcomes. Thirteen studies includedcondition-specific outcomes including PND duration[33]; cases recovered or improved [28, 38, 39], casesdetected or averted [27, 28]; improvement in PNDsymptoms [26, 29, 34, 41] or scores from a screeningtool [31, 32, 35]. Five studies also used generic out-comes such as well-being [26, 30, 34] and generalhealth [35, 41]. Six studies measured health in termsof QALYs from the maternal perspective [28, 36–38,40, 42]. However, health outcomes relating to childrenwere considered in only four studies, this was interms of cognitive functioning [30], sleep [26], tem-perament [26, 41], and depression [34]. Seven studiesacknowledged that outcomes for children and/or partnerhealth were important or likely to be affected but did notinclude them in their analysis [28, 36–40, 42]. Only threeof the seven studies explained their omission; this was typ-ically due to a lack of reliable data [28, 36], or due to miss-ing data [37]. There was no mention of child or partner/family health outcomes in four studies [27, 29, 32, 33].Similarly, non-health outcomes were explicitly considered

by four studies only in relation to PND [29, 30, 34, 41].Nearly all of these studies included outcomes relating tosocial or emotional support for PND [29, 30, 41].Other non-health outcomes included mother’s em-ployment and earnings, parent-infant interaction, chil-dren’s educational attainment and behavioural problems,couples’ relationships, satisfaction and efficacy in parent-ing role, and family functioning. Another four studies ex-plicitly acknowledged the significance of non-healtheffects, but did not include them [26, 28, 38, 39]. A lack ofrelevant evidence was the main reason stated for exclud-ing potential non-health effects [30, 34, 38].

Outcome measurement and valuationDifferent instruments were used to assess the presence,risk or duration of PND as an outcome in the economicevaluation (Tables 2 and 3). Seven studies employed theEdinburgh Postnatal Depression Scale (EPDS) [26, 27,

31, 32, 35, 38, 41]. However, although the same tool wasused, a range of thresholds were used. For example, thethresholds used to define the risk of PND ranged from 9above [26] to 13 or above [31]. Some defined the EPDSthreshold in relation to its level of specificity and sensi-tivity, by validating it against an existing diagnostic tool[31] or by piloting it on trial participants [32], whileothers referred to different published sources [26, 27].Other instruments were also used, with three studiesemploying the Patient Health Questionnaire [28, 29, 38],two using the Structured Clinical Interview for Depression[27, 33] and one study adopting the Clinical InterviewSchedule [39].In all the economic models that used QALYs, the util-

ity weights were derived from secondary sources(Table 3). Three studies used the same source for utilityvalues [28, 36, 42]. In most cases, the utility values usedwere based on the health states associated with de-pressed or general populations rather than women withPND. For other studies involving a monetary valuationof outcomes, some of the valuations of outcomes werebased on authors’ own estimates, due to a lack of avail-able data [30].

Other methodological considerationsStudy perspectiveThe most common perspective adopted by the economicevaluations was a National Health Service/PersonalSocial Services (NHS/PSS) or a healthcare perspectiveonly (n = 10) [26, 28, 31–33, 36–38, 40, 41]. Three stud-ies adopted a societal perspective only [34, 39, 42]. Twostudies took a societal perspective alongside other per-spectives such as a public sector perspective [30], athird-party payer perspective, a healthcare perspectiveand family perspective [27]. In one study [35], a patientperspective was taken alongside a healthcare perspective.

Time horizon and discountingThere were some variations in terms of the time hori-zon adopted for costs and consequences by the eco-nomic evaluations (Table 4). The most common timehorizon for outcomes was a year (n = 9), followed bysix months (n = 3), 18 months (n = 2), and 12 weeks(n = 2). Only two studies adopted time horizons lon-ger than a year, justifying them as necessary due tothe short and longer-term impacts of PND [29, 33].Justifications for adopting a limited time horizon in-cluded constraints associated with the trial follow-upperiod [27, 33], practical limitations and budget con-straints [39]. As most studies had the time horizon ofa year or less, discounting of benefits was not re-quired. The two studies adopting a longer time period

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Table

2Descriptio

nof

outcom

esused

inRC

Tor

coho

rt-based

econ

omicevaluatio

ns

Stud

y(Year)

Interven

tion

Outcomes

Outcomes

othe

rthan

maternaland

health

outcom

es

How

was

theou

tcom

emeasuredand/or

valued

?

Source

Otheroutcom

esmeasuredinthe

trialbutn

otused/m

aybe

relevant

intheeconom

icevaluation

Outcomes

acknow

ledg

edbu

texclud

ed

Boath

(2003)

[39]

PBDUcustom

ised

treatm

ent

•PN

Dcasesrecovered

–CIS

Coh

ort

stud

y•Anxiety

•Workleisureandfamily

life

•Marriage

quality

andothe

rsimilardyads

Child

andno

n-he

alth;reasons

for

theirno

n-inclusionno

tprovided

Dukho

vny

(2013)

[27]

Teleph

one-basedpe

ersupp

ort

interven

tion,

access

tostandard

postpartum

care

•PN

Dcasesaverted

–EPDS(thresho

ldof

≤12

forlow

risk),SCID

RCT

•Anxiety

•Lone

liness

•Satisfactionwith

interven

tion

Hiscock

(2007)

[26]

Individu

alstructured

maternaland

child

health

consultatio

ns,a

choice

ofbe

haviou

ralinterventions,

‘con

trolledcrying

’or‘cam

ping

out’

•Depressionsymptom

s•Men

taland

physicalhe

alth

scores

•Sleepqu

ality

andqu

antity

•Infant’ssleepprob

lem

(prim

aryou

tcom

e)•Infant’stempe

ramen

t

Child

EPDS(thresho

ld>9for

PND),SF-12,sleep

questio

ns,night

waking

indicator,GlobalInfant

Tempe

ramen

tScale

RCT

NA

MacArthu

ra

(2003)

[31]

Rede

sign

edmod

elof

commun

itypo

stnatalcare(m

idwifery-led)

•PN

Dscore

–EPDS(score

of≥13

indicatedrisk)

RCT

•PhysicalandMentalH

ealth

•Repo

rted

morbidity

•‘Goo

dpractice’

•Wom

enandprofessionalsview

saboutcare

Child;reasons

forits

non-inclusion

notdiscussed

Morrella

(2000)

[32]

Com

mun

itymidwifery

supp

ortworkers

•PN

Dscore

–EPDS(score

of≥12

indicatedrisk)

RCT

•Generalhealth

perception

•Functio

nalSocialSup

port

•Breastfeed

ing

Morrell

(2009)

[37]

Health

visitortraine

dto

iden

tify

andde

liver

CBA

orpe

rson

-cen

tred

approach

(listen

ingvisits)

•QALY

–TheSF-6D,from

asubset

ofSF-36qu

estions,w

ascalculated.SF-6D

scores

estim

ated

usingUKtariffs.

RCT

•Prop

ortio

nof

at-riskwom

en(prim

ary)

•EPDSscore

•Ph

ysicalandmentalhealth

•ClinicalOutcomes

inRo

utine

Evaluatio

n•Anxiety

•Perceivedstressfulimpact

ofhaving

ayoun

gchild

•Cou

ples

relatio

nship

•Co

gnitive,socialand

emotionaldevelop

mentof

infants

•Risk

ofdeveloping

autism

Child,p

artner/fam

ily;the

seou

tcom

escouldno

tinclud

eddu

eto

missing

data

Petrou

(2006)

[33]

Cou

nsellingandsupp

ortpackage

bytraine

dhe

alth

visitors

•Durationof

PND

expe

rienced

–SC

ID-II

RCT

Unclear

whatothe

rou

tcom

esweremeasuredin

thetrial

Price

(2015)

[29]

Enhanced

engage

men

tin

home

visitingviamotivationalinterview

ing

andbriefintervention(CBT

and

InterpersonalTherapy)

•Dep

ressivesymptom

s•Socialsupp

ort

Non

-health

Patient

Health

Questionn

aire-9,

Sarason’sSocialSupp

ort

Questionn

aire-Revise

d

Coh

ort

stud

yNA

Sembi

(2016)

[41]

Teleph

onepe

ersupp

ort

•Dep

ressivesymptom

s•Parent-in

fant

interaction

•Anxiety

•Em

otionalsup

port

Child,non

-health

EPDS(score

of>9or

10indicatedmild

depressio

n),

CARE-Index,Hospital

Anxietyan

dDep

ression

RCT

NA

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Table

2Descriptio

nof

outcom

esused

inRC

Tor

coho

rt-based

econ

omicevaluatio

ns(Con

tinued)

Stud

y(Year)

Interven

tion

Outcomes

Outcomes

othe

rthan

maternaland

health

outcom

es

How

was

theou

tcom

emeasuredand/or

valued

?

Source

Otheroutcom

esmeasuredinthe

trialbutn

otused/m

aybe

relevant

intheeconom

icevaluation

Outcomes

acknow

ledg

edbu

texclud

ed

•Parents’satisfactionand

efficacyin

theirparenting

role

•Optim

istic

self-be

liefs

•Cou

ples

relatio

nship

•Infant

tempe

ramen

t•Percep

tions

ofinterven

tions

•Gen

eralhe

alth

Scale,Em

otionalSup

port

Questionn

aire,Parentin

gSenseof

Competence

scale,Generalise

dSelf-

efficacyQuestionn

aire,

DyadicAdjustm

entScale,

Infant

Temperament

Questionn

aireandPeer

Supp

ortEvaluation

Inventory,SF-12

Wiggins

a

(2004)

[35]

Health

visitorsupp

ortor

Com

mun

itygrou

psupp

ort

•PN

Dscore

–EPDS(score≥12

indicated

high

risk)

RCT

•Child

injury

•Maternalsmoking

•Socialsupp

ort

•Maternaland

child

health

•Infant

feed

ing

•Mothe

r-child

interaction

•Hou

seho

ldresources

CBACog

nitiv

eBe

haviou

ralA

pproach,

CBTCog

nitiv

eBe

haviou

ralT

herapy

,ClSClin

ical

interview

sche

dule,EPD

SEd

inbu

rghPo

stna

talD

epressionScale,PB

DUPa

rent

andba

byda

yun

it,RC

TRa

ndom

ised

Con

trolledTrial,SC

IDStructured

Clin

ical

Interview

forDep

ression,

SFSh

ortFo

rm,N

ANot

applicab

lea Study

focusedon

PNDan

dothe

raspe

cts–othe

rou

tcom

esused

inthetrialm

ayno

tne

cessarily

relate

toPN

D

Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 9 of 18

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Table

3Descriptio

nof

outcom

esused

inmod

el-based

econ

omicevaluatio

ns

Stud

y(Year)

Interven

tion

Outcomes

Outcomes

othe

rthan

maternaland

health

outcom

es

How

was

the

outcom

emeasured

and/or

valued

?

Source

Keyassumptions

Outcomes

acknow

ledg

edbu

texclud

ed

Battye

(2012)

[30]

Befrien

ding

service

(telep

hone

helplineand

one-to-one

supp

ortby

traine

d‘befriend

er’

volunteers)

Short-term

•Im

provem

entsin

men

tal

health

•Increasedaw

aren

essof

PND

andPN

Dsupp

ort

•Increasedcoping

ability

Long

-term

•Redu

cedinfantsbe

haviou

ral

prob

lems

•Im

proved

infantscogn

itive

functio

ning

•Family

functio

ning

improvem

entH

ealthcare

professio

nalsandvolunteers

outcom

esalso

measured.

Child,othersaand

non-he

alth

ShortWarwick-

Edinbu

rghMen

tal

Wellbeing

Scale,

qualitativeinterviews

andevaluatio

nform

Questionn

aires,

qualitativeinterviews,

mon

itorin

gdata,and

publishe

dstud

ies

Interven

tionbe

nefits

willsustainin

the

future

with

only20%

drop

-off.

Bauer(2011)

[42]

Universalhe

alth

visitin

g(postnatalscreen

ing

usingEPDSand

treatm

ent[CBT+

antid

epressant])

•QALY

–Utilities

for

depression

states

derived

from

second

arysources.

Benn

ettet

al.[51]

andRevickiand

Woo

d[52]

With

outtreatm

ent,

PNDwillsustainwith

ashort-term

resolution.

Symptomsof

mod

erate-to-severe

PND

arecomparab

leto

thoseof

mod

erate-

to-severedepressio

n.

Child

andno

n-he

alth;

reason

sfortheirno

n-inclusionno

tprovided

Cam

pbell(2008)[28]

Routinescreen

ing

prog

ramme(usin

gPH

Q-2)

andtreatm

ents

(antidep

ressants,

psycho

logicaltherap

ies

orsocial

supp

ort)

accordingto

severity

ofPN

D.

•PN

Dcasesde

tected

•PN

Dcasesresolved

•QALY

–PH

Q-2,Preference

weigh

tsforQALYs

derived

from

asecond

arysource.

Second

arysources,

Revickiand

Woo

d[52]

Normalutility

six-

weeks

post-treatmen

tin

thetreatm

ent

respon

ders.N

on-

respon

derswith

mild/m

oderate

depression

recover

with

insixmon

thsof

itson

set.PN

Dwill

sustainin

unde

tected

casesandno

n-respon

derswith

severe

depression

.Alinearde

terio

ratio

nor

improvem

entb

etween

health

states

overtim

e.

Child

andno

n-he

alth;

child

outcom

escould

notbe

includ

eddu

eto

lack

ofreliable

data

Hew

itt(2009)

[36]

Iden

tification

1.EPDS

2.Beck

Dep

ression

Inventory

Treatm

ents

1.Structured

psychological

therapy

2.Listen

ingvisit

•QALY

–Utility

weigh

tsde

rived

forQALYs

from

asecond

ary

source.

Effectiven

essestim

ate

from

asystem

atic

review

andmeta-

analysis,utility

values

from

Revickiand

Woo

d[52]

Non

-respo

ndersto

treatm

entandusual

care

wou

ldremain

depressedun

tilthe

mod

elen

dpoint.

Wom

enen

terthe

relevant

treatm

entat

6weeks

postnatally.

Child

andpartne

r/family;these

outcom

escouldno

tbeinclud

eddu

eto

lack

ofreliable

data

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Table

3Descriptio

nof

outcom

esused

inmod

el-based

econ

omicevaluatio

ns(Con

tinued)

Stud

y(Year)

Interven

tion

Outcomes

Outcomes

othe

rthan

maternaland

health

outcom

es

How

was

the

outcom

emeasured

and/or

valued

?

Source

Keyassumptions

Outcomes

acknow

ledg

edbu

texclud

ed

(Bothwith

preced

ing

additio

nalcare)

Alinearde

terio

ratio

nor

improvem

ent

betw

eenhe

alth

states

over

time.

NCCMH(2014)

[38]

Iden

tification

1.EPDSon

ly2.Who

oley

questio

nsfollowed

byEPDS

3.Who

oley

questio

nsfollowed

byPH

Q-9

Treatm

ent

1.Facilitated

self-he

lpbasedon

CBT

principles

2.Listen

ingvisits

(Bothin

additio

nto

standard

postnatalcare)

Iden

tification

•QALY

Treatm

ent

•QALY

•PN

Dcasesim

proved

andno

trelapsed

–EPDS,Who

oley

questio

n,PH

Q-9.U

til-

ityweigh

tsde

rived

forQALYsfro

masec-

ondary

source.

Effectiven

essestim

ate

from

meta-analyses,

utility

values

from

Sapinandcolleagues

[53],expertsop

inion

Iden

tification

Falsene

gative

wom

encouldhave

spon

tane

ous

recovery

orbe

iden

tifiedin

theGP

follow-upandoffered

treatm

ent.Onlyfirst-

linetreatm

entscon-

side

redandrelapse

notmod

elled.

Treatm

ent

Wom

enwho

improve

remaininthestateor

relapseun

tilthe

mod

elendp

oint.A

lineard

eterioratio

nor

improvem

ent

betweenhealth

states

over

time.

Child,p

artner/fam

ilyandno

n-he

alth;

reason

sforexclud

ing

non-he

alth

outcom

eswasthelack

ofrelevant

evidence

Steven

son(2010)

[40]

Group

CBT

•QALY

–Chang

esin

EPDS

scores

were

translated

tochange

sin

utility

using

second

arydata.

Datafro

mMorrellet

al.[37]

Bene

fitswou

ldsustainover

the6-

mon

thpe

riodwith

lineard

eclineafterwards

tozero,a

yearafterthe

treatment.

Child

andpartne

r/family;reasons

for

theirno

n-inclusion

notprovided

Taylor

(2014)

[34]

Socialsupp

ort

(e.g.advocacy,

befrien

ding

)

•Increasedwell-b

eing

•Increasedchancesof

employmen

tandhigh

erearnings

•Long

-term

bene

ficialchildren

outcom

es•Redu

ceduseof

health

and

socialcare

services

•Increasedtaxrevenu

es•Vo

lunteersbe

nefits

Child,othersaand

non-he

alth

HospitalA

nxiety

and

Dep

ressionScale,

analysisof

acoho

rtstud

y

Expe

rts,arang

eof

second

arysources

Bene

fitswere

estim

ated

from

anob

servationalstudy

andan

RCTof

similar

service.Bene

fitsfor

wom

enandsociety

inferred

from

expe

rts

andarang

eof

publishe

dstud

ies.

CBTCog

nitiv

eBe

haviou

ralT

herapy

,EPD

SEd

inbu

rghPo

stna

talD

epressionScale,

NCC

MHNationa

lCollaboratingCen

treforMen

talHea

lth,

PHQ

Patien

tHea

lthQue

stionn

aire,QALY

Qua

lity-ad

justed

-life-year,RC

TRa

ndom

ised

ControlledTrial

a Othersinclud

epa

rtne

r/family,volun

teersor

healthcare

profession

als

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Table

4Metho

dologicalcon

side

ratio

nsandcost-effectiven

essresults

Lead

author

(Year)

Interven

tion

Perspe

ctive(re

ason

s)Timeho

rizon

used

inecon

omic

evaluatio

n(re

ason

s)

Discoun

ting

Keycost-effectiven

essresults

RCTor

coho

rt-based

econ

omicevaluatio

ns

Boath

(2003)

[39]

PBDUcustom

ised

treatm

ent

Societal

6mon

ths(practical

considerations,

budg

etaryconstraints)

Costs:6%

“The

curren

ttreatm

entof

postnatald

epressionisdo

minated

onthegrou

ndsof

cost-

effectiven

essby

PBDU

treatm

ent.Themovefro

mRPCto

PBDUwou

ldincuran

additio

nalcostexpe

nded

per

successfullytreatedwom

anof

£1945.”

Dukho

vny

(2013)

[27]

Teleph

one-basedpe

ersupp

ortinterven

tion,

access

tostandard

postpartum

care

Societal(USand

Canadiangu

idelines)

Third

-party

payer,

Health

care,Fam

ilype

rspe

ctive

12weeks

(RCTtim

eho

rizon

)No**

Theinterven

tionwas

foun

dto

becost-effective.

ICER:

CAD$10,009pe

rcase

ofPN

Daverted

Thereis95%

prob

ability

that

theprog

ram

wou

ldcostless

than

CAD$20,196pe

rPN

Dcase

averted

Hiscock

(2007)

[26]

Individu

alstructured

maternaland

child

health

consultatio

ns,

achoice

ofbehaviou

ral

interventions,‘con

trolled

crying

’or‘camping

out’

NHS/PSS*

10,12mon

ths

No

Bene

fits

Infant

sleepprob

lems

At10

mon

ths,56%

ofinterven

tionand68%

ofcontrolm

othe

rsrepo

rted

infant

sleep

prob

lems(OR0.61,p

=0.04);At12

mon

ths,thisfellto

39%

vs55%

(OR0.53,p

=0.007).

EPDSscores

Interven

tionmothe

rshadlower

meanEPDSscores

than

controlsat

12mon

ths(5.9vs

7.2,p=0.001)

andhigh

ermen

talh

ealth

(SF-12)scores

atbo

th10

mon

ths(48.1vs

45.0,

p=0.001)

and12

mon

ths(49.7vs

46.1,p

=0.001).

Costs

Interven

tion:£96.93

(SD,£249.37)

Con

trol:£116.79

(SD,£

330.31)

Meandifference:£19.44

(95%

CI£283.70

to£44.81,p

=0.55)

MacArthu

r(2003)

[31]

Rede

sign

edmod

elof

commun

itypo

stnatal

care

(midwifery-led)

Health

care

12mon

ths

No**

“The

cost-con

sequ

encesanalysisestablish

edthatthecostsof

theinterventio

nandcontrol

care

werebroadlyequivalent.The

interventio

ncare

costingatamaximum

£81.90

more

perw

oman

todeliver,but

possiblyrepresentin

gasaving

of£78.30

perw

oman,depending

onassumptions

used.”

Morrell

(2000)

[32]

Com

mun

itymidwifery

supp

ortworker

Health

care

6weeks

(Valid

useof

EPDS

enablingcomparability

with

othertrials)

Costs:5%

Given

thathealth

outcom

esweresim

ilarfor

both

grou

ps,the

econ

omicanalysisislim

ited

toacomparison

ofcostsbetweentheinterventio

nandcontrolgroup

s.Meantotalcostto

theNHSat

6weeks

(prim

aryanalysis)

Interven

tiongrou

p:£635

(SD,£326)

Thecontrolg

roup

:£456(SD,£291)

Meandifference:£180

(95%

CI,£126,£232,p=0.001).

Morrell

(2009)

[37]

Health

visitor

trainedto

identifyanddeliverCB

Aor

person

-centred

approach

NHS/PSS(NICE

guidelines)

6mon

ths

No**

Theinterventiondo

minated

thecomparatorfor

at-risk

wom

enat6mon

ths(prim

aryanalysis).

How

ever,a

significantd

ifference

was

noto

bservedinthenu

mbero

fQALYsgained

inthe

interventiongroups

comparedto

thecontrolgroup

andtherewas

uncertaintyassociated

with

thecostandQALYpairs.The

prob

abilityofCB

Tbeingcost-effectivewas

justover70%***

Petrou

(2006)

[33]

Counsellin

gandsupp

ort

packageby

trained

health

visitors

Health

care

18mon

ths

(RCTtim

epe

riod)

Outcomes:1.5%

Costs:6%

Theinterventioniscost-effectivecomparedto

RPC.

ICER

£43.1pe

rmon

thof

PNDavoide

d.Theprob

ability

that

theinterven

tioniscost-effectiveexceed

s70%

once

decision

makers

expressawillingn

essto

invest£1000to

preven

teach

mon

thof

PND.

Price

(2015)

[29]

Enhanced

engagement

inho

mevisitingvia

motivationalinterview

ing

Serviceproviders*

12weeks

No

Bene

fits

Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 12 of 18

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Table

4Metho

dologicalcon

side

ratio

nsandcost-effectiven

essresults

(Con

tinued)

Lead

author

(Year)

Interven

tion

Perspe

ctive(re

ason

s)Timeho

rizon

used

inecon

omic

evaluatio

n(re

ason

s)

Discoun

ting

Keycost-effectiven

essresults

andbriefintervention

(CBT

andInterpersonal

Therapy)

“Ade

crease

inde

pressive

symptom

sassociated

with

theinterven

tionthat

approached

statisticalsign

ificance(p=0.0600).Sign

ificant

increase

inpe

rceivedsocialsupp

ort

(t=3.35,p

=0.0027).”

MeanCosts

Usualcare:$158.30

perp

articipant

Enhanced

Engage

men

t:$147.50pe

rparticipant

Sembi

(2016)

[41]

Teleph

onepeersupp

ort

Health

care

6mon

ths

No

Bene

fits(prim

aryou

tcom

es)

Nosig

nificantdifferences

between-subjectsandimprovem

entinmother-infant

interaction.

Costs

Meancostof

thecombine

duseof

NHSresources

Fortheinterven

tiongrou

p:£800.67(SD,£761.74)

Forstandard

care

grou

p:£1537.80

(SD,£1936.37).

Itwas

notp

ossib

leto

cond

ucta

cost-effectivenessanalysisdueto

thesm

allnum

bero

fpatients.

Wiggins

(2004)

[35]

Health

visitorsupp

ort

orCom

mun

itygrou

psupp

ort

Health

care,Patients

12,18mon

ths

6%(costs)

Bene

fits

Therewas

nocleardifferencein

anyof

theprim

aryou

tcom

es.

Maternaldepression:Fewer

wom

eninthecombinedinterventiongrou

pscored

over

the

depressio

nthresholdon

theEPDS(−3%

)thanthecontrolgroup

MeanCosts

TheSupp

ortHealth

Visitorinterven

tionem

erge

das

arelativelyexpe

nsiveinterven

tion

toim

plem

entcomparedwith

theCom

mun

ityGroup

Supp

ortinterven

tion.

Supp

ortHealth

Visitor=

£3255(SD,£2253)

Com

mun

ityGroup

Supp

ort:£3231(SD,£3323)

Controlgroup:£2915

(SD,£2349)

Mod

el-based

econ

omicevaluatio

ns

Battye

(2012)

[30]

Befrien

ding

service

(telep

hone

helpline

andon

e-to-one

supp

ortby

traine

d‘befriend

er’volun

teers)

Societal,p

ublic

sector

(dem

onstrate

valueto

societyandhe

althcare)

Outcomes

3,6and30

years

Costs

1year

3.5%

Thebe

frien

ding

servicewas

cost-ben

eficialtobo

thsocietyandthestate.

Societalpe

rspe

ctive

Forevery£1

invested

,the

estim

ated

SROI:

▪£3

over

theshortterm

▪£4

over

themed

ium

term

▪£6.50over

thelong

erterm

Publicsector

perspe

ctive

Forevery£1

invested

,the

estim

ated

SROI:

▪£0.20over

theshortterm

▪£0.20over

themed

ium

term

▪£1.50over

thelong

erterm

Bauer

(2011)

[42]

Universalhealth

visiting

(postnatalscreening

usingEPDSandtreatment

[CBT

+antidepressant])

Societal*

12mon

ths

No

Health

visitin

ginterven

tionprovided

apo

sitivene

tbe

nefit.

ICER

£4500pe

rQALY

gained

Net

mon

etarybe

nefits

£640

permothe

r(atWTP

thresholdof

£20,000).

Byextrapolation,thisam

ountsto

arou

nd£300

millionforEngland.

Cam

pbell

(2008)

[28]

Routinescreen

ing

prog

ramme(using

PHQ-2)andtreatm

ents

Health

care

12mon

ths

No**

Theprop

osed

routinescreeningprog

rammeappearsto

behigh

lycost-effectivecompared

tothecurrentpracticefro

mago

vernmentperspective.

ICERs

•NZ$287

peradditio

nalcasede

tected

Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 13 of 18

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Table

4Metho

dologicalcon

side

ratio

nsandcost-effectiven

essresults

(Con

tinued)

Lead

author

(Year)

Interven

tion

Perspe

ctive(re

ason

s)Timeho

rizon

used

inecon

omic

evaluatio

n(re

ason

s)

Discoun

ting

Keycost-effectiven

essresults

accordingto

severity

ofPN

D.

•NZ$400

peradditio

nalcaseresolved

•NZ$3461pe

radditio

nalQ

ALY

gained

Hew

itt(2009)

[36]

Iden

tification

1.EPDS

2.BD

ITreatm

ents

1.Structured

psycho

logicalthe

rapy

2.Listen

ingvisit

(Bothwith

preced

ing

additio

nalcare)

NHS/PSS

12mon

ths

No**

Iden

tification

ICER

Theiden

tificationstrategies

wereno

tcost-effectivecomparedto

thecurren

tpractice.

EPDSat

acutpo

intof

16:3£41,204pe

rQALY

gained

.Other

cutpo

intsandBD

Icut

point10

wereeither

dominated

orhadICERshigh

erthan

that

ofEPDScutpo

int16.

Ateach

ofthethreeWTP

thresholds

considered

(£20,000,£30,000

and£40,000),the

strategy

with

thehigh

estind

ividualprobabilityof

beingcost-effectivewas

routinecase

detection.

Treatm

ent

Structured

psycho

logicaltherapy

was

acosteffectivetreatm

ent**bu

tlistening

homevisits

was

notcost-effectivecomparedto

thecurrentpractice.

ICER

Structured

psychologicaltherapy:£17,481

perQ

ALYgained

Listen

ingho

mevisits:£66,275

perQALY

gained

Therewas

50%

prob

ability

that

structured

psycho

logicalthe

rapy

wou

ldbe

cost-effective

ataWTP

thresholdof

£20,000pe

rQALY

gained

.

NCCMH

(2014)[38]

Iden

tification

1.EPDSon

ly2.Who

oley

questio

nsfollowed

byEPDS

3.Who

oley

questio

nsfollowed

byPH

Q-9

Treatm

ent

1.Facilitated

self-he

lpbasedon

CBT

principles

2.Listen

ingvisits

(Bothin

additio

nto

standard

postnatalcare)

NHS/PSS(NICE

guidelines)

Iden

tification

12mon

ths

Treatm

ent

12mon

ths7weeks

No**

Iden

tification

The‘Who

oley

questio

ns’followed

byPH

Q-9

was

estim

ated

tobe

themostcost-effective

iden

tificationstrategy,how

ever,w

ellabo

vetheNICEthreshold**.

ICER

Who

oley

questio

nsfollowed

byEPDSversus

Who

oley

questio

nsfollowed

byPH

Q-9:

£45,593pe

rQALY

gained

Treatm

ent

Facilitated

self-helpcomparedwith

standard

carewas

overallm

oreeffectiveandmorecostly.

ICER

Facilitated

self-help:£2269

peradd

ition

alwom

animprovingandno

trelapsingattheend

ofthemod

el,or£

13,324

perQ

ALY

gained.

Theprob

ability

offacilitated

self-he

lpbe

ingcosteffectiveis0.59

to0.72***.

Steven

son

(2010)

[40]

Group

CBT

NHS/PSS

(NICEgu

idelines)

12mon

ths

No**

Thegrou

pCBT

comparedwith

RPCwas

notfoun

dto

becost-effective.***

ICER

CBT:£46,462

perQALY

gained

(95%

CI,£37,008to

£60,728).

Taylor

(2014)

[34]

Socialsupp

ort(e.g.

advocacy,b

efriend

ing)

Societal*

(tode

term

inevalueto

society)

12mon

ths-over

alifetim

e3.5%

(outcomes)

Estim

ated

averagedirectfinancialcostofprovidingsupp

ort:£2230perw

oman.

Estim

ated

bene

fitUsing

SF-6D:£591–£887

perwom

antreated

Using

EQ-5D:£1302–£1954

perwom

antreated

*Not

explicitlystated

byau

thors

**Re

ason

sprov

ided

:due

tosm

alltim

eho

rizon

***A

tawillingn

ess-to-pay

thresholdof

£20,00

0-£3

0,00

0/QALY

gained

BDIB

eckDep

ressionInventory,CA

DCan

adian,

CBACog

nitiv

eBe

haviou

ralA

pproach,

CBTCog

nitiv

eBe

haviou

ralT

herapy

,EPD

SEd

inbu

rghPo

stna

talD

epressionScale,

EQ-5DEu

roQol-5

dimen

sion

s,ICER

Increm

ental

cost-effectiv

enessratio

,NCC

MHNationa

lCollabo

ratin

gCen

treforMen

talH

ealth

,NHSNationa

lHealth

Service,

NZNew

Zealan

d,OROdd

sratio

,PBD

UPa

rent

andba

byda

yun

it,PH

QPa

tient

Health

Que

stionn

aire,P

SSPe

rson

alSo

cial

Services,Q

ALY

Qua

lity-ad

justed

-life-year,RC

TRa

ndom

ised

Con

trolledTrial,RP

CRo

utineprim

arycare,SDStan

dard

deviation,

SF-6DSh

ortFo

rm-6dimen

sion

s,SROIS

ocialreturnon

investmen

t,WTP

Willingn

ess-to-pay

Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 14 of 18

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discounted the benefits appropriately at the recom-mended rate [30, 34].

Intermediate outcomesSome studies used intermediate outcomes such as casesdetected or averted [27, 28]. While such outcomes maybe meaningful in the context of PND interventions, theycan be of more limited general use for commissioners asthey do not allow comparison of cost-effectivenessacross programme areas [10].

Sensitivity analysisAlmost half of the studies did not explore uncertaintyaround the estimates of outcomes within a sensitivityanalysis. Those studies that performed sensitivity ana-lysis mainly conducted a deterministic sensitivity analysis[28, 30, 32, 39, 40, 42] and a few studies conducted prob-abilistic sensitivity analysis [27, 38, 40].

Cost-effectiveness of interventionsOf the 11 studies conducting full economic evaluations,10 reported that the intervention under investigation ap-peared to be cost-effective (Table 4). Of those 10 studies,three studies found that a combination of PND screen-ing and treatment was cost-effective [28, 37, 42], a fur-ther three studies reported that treatments such aspsychological therapy, facilitated self-help and custom-ized treatment were more cost-effective than standardcare [36, 38, 39], and four studies found positive resultsfor preventive strategies which involved peer support orcounselling and other specific support [27, 30, 33, 34].Group CBT was not found to be cost-effective comparedto standard care in one study [40].

DiscussionThe systematic review identified 17 studies of interven-tions to prevent and/or treat PND, in which an eco-nomic evaluation was conducted. The majority of thestudies focused on psychological or psychosocial interven-tions and none focused on pharmacological interventionsonly. Overall, 10 of the 11 full economic evaluations re-ported that an intervention was cost-effective. These in-volved a variety of interventions ranging from additionalsupport from health professionals, peer support and com-bined screening and treatment strategies which were usu-ally compared with standard care. The review identified anumber of methodological issues relating to how out-comes were included, measured and valued in the eco-nomic evaluations; these related to whose outcomes wereincluded, the inclusion of relevant health and non-healthoutcomes, study perspective and time horizon.Guidelines emphasise the need to identify all relevant

outcomes in an economic evaluation [25, 43]. However,only four studies considered health outcomes associated

with children [26, 30, 34, 41]. This raises concerns sincenumerous studies have shown the adverse impacts ofPND on the child’s health and development, and ontheir interaction with their mothers [20, 44]. The exclu-sion of children’s outcomes from an economic evalu-ation may mean that an incomplete assessment ofcost-effectiveness has been undertaken. For example, anintervention found to be less cost-effective compared toanother intervention on the basis of maternal outcomesonly, may well be more cost-effective when potentialbenefits to the infant’s health are included. However,there could be potential barriers to considering infants’outcomes such as lack of robust data, or an inability tomeasure outcomes directly for children. A further meth-odological barrier could be related to concerns about in-creasing the likelihood of findings of false significance(type I error) due to the inclusion of multiple outcomesin an evaluation. Similarly, health outcomes for thefather and wider family are potentially relevant and re-quire consideration [19, 21].Non-health outcomes are relevant and important in

the context of PND [18]. However, presently, there is noaccepted method to determine which non-health effectsare important and how they should be incorporated inan economic analysis [43]. A range of potentialapproaches for public health interventions have beenoutlined that allow for the inclusion of health and non-health outcomes (e.g. cost-consequence analysis, cost-benefit analysis etc.) [21]. The focus of most of thestudies was exclusively on health, with only four studiesmeasuring some kind of non-health outcomes. Severalauthors deemed non-health outcomes to be importantbut did not include them in their evaluation due to chal-lenges such as a lack of reliable and quantifiable data,missing data, and more than one primary outcome beingincluded in the trial.Some methodological issues were evident relating to

the measurement and valuation of outcomes. A detailedanalysis of the properties and limitations of the existinginstruments used to capture outcomes is essential to in-form appropriate ways to measure those outcomes. Forexample, the frequently used EPDS tool had various cut-off thresholds, indicating differing approaches to usingthis tool. Many authors of the included studies alsomentioned the lack of reliable data on utilities. For ex-ample, Stevenson et al. [40] used regression techniquesto estimate utilities (based on data from a different trial)but acknowledged that this introduced further uncer-tainty in the analysis. Other authors used utility weightsbased on the health states associated with general de-pression and not PND. Although PND and general de-pression share some similar symptoms they differ incertain characteristics such as the experience of child-birth and sleep deprivation [44]. If utility estimates do

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not directly relate to PND, there is a possibility thatthe utilities may overestimate or underestimate theintervention effects.A societal perspective is generally considered the most

appropriate perspective for PND interventions due tothe wide range of impacts associated with the condition[21, 45, 46]. This is in keeping with guidance relating tothe economic evaluation of public health interventions,where a perspective broader than the healthcare per-spective may be necessary [21]. This would enable out-comes beyond health to be considered, such as thoserelating to education, housing, crime etc. However, theresults of this review demonstrated that only five studiesadopted a societal perspective.Another recurrent issue observed was the limited time

length adopted by most of the studies. The time horizonwas no more than 18 months in the trial-based eco-nomic evaluations. It can be argued that important dif-ferences between the interventions may not be capturedusing short time-horizons. For example, a prospectivelongitudinal study showed that the children, who wereadversely affected in their infancy due to their mother’sPND at 3 months postpartum, experienced more prob-lems with intellectual and academic performance at11 years of age compared to the children of healthymothers [20] and those problems could have potentialeconomic consequences such as additional school sup-port costs and productivity losses from leaving schoolwithout qualifications [10]. Thus, studies adopting a lon-ger time horizon are needed to be able to capture thelong-term effects of PND.This review has several strengths. Systematic and

rigorous processes were adopted to identify and assessstudies. A comprehensive search strategy was imple-mented which also included searches for unpublished re-ports. Both prevention and treatment strategies wereincluded, providing a holistic overview of several meth-odological issues concerning outcome identification andmeasurement for the economic evaluation of PND inter-ventions. Using established criteria [25] a qualityappraisal process was undertaken analysing all key ele-ments relating to outcomes.Nonetheless, the review is subject to some limita-

tions. Firstly, potential studies may have been missedby the search strategy either due to inadequate classifi-cations of economic terms in the databases or due tothe different ways interventions to improve mentalhealth in the postpartum period can be coded depend-ing on the type or the focus of intervention (e.g. on themother, the infant etc.) [47]. Secondly, since we couldnot find detailed guidelines focussed on economic eval-uations of PND interventions, our analysis of the qual-ity of the studies was based on generic guidance. Lastly,an in-depth analysis of evidence on the clinical and

cost-effectiveness of the interventions was beyond thereview’s scope.This is the first systematic review to examine the ap-

proaches taken and types of outcomes used in economicevaluations of PND interventions (for prevention andtreatment). A systematic review undertaken by theNational Collaborating Centre for Mental Health [38]was concerned with accumulating evidence on the cost-effectiveness of interventions to prevent or treat mentalhealth problems in pregnancy and the postnatal period.More recently a systematic review [16] was conducted toinform parameters for a model-based economic evalu-ation of antenatal and postnatal interventions for preg-nant and postnatal women to prevent PND. While thesestudies attempted to identify economic evaluations ofPND interventions, they did not explore the methodo-logical issues associated with the approaches taken andoutcomes adopted in the studies.The findings of this review highlight several implica-

tions for future research. Future economic evaluationsshould identify and consider the full range of potentialoutcomes that are relevant in the context of PND: healthand non-health outcomes, maternal, family and childoutcomes. The development of new methods and refine-ment of existing approaches that can incorporate bothhealth and non-health benefits of intervention are essen-tial for a complete evaluation of the cost-effectiveness ofPND interventions. The list of outcomes generated fromthis review, as a preliminary framework, could be refinedfurther through engagement with key stakeholders in-cluding mothers, family members, clinicians and health-care commissioners to reach consensus on whatoutcomes are important for use in economic evaluationsof PND interventions and in wider evaluations of inter-ventions and services. Given the wide range of impactsassociated with PND, in order to allow a full assessmentof costs and consequences, a societal perspective shouldbe considered. However, key challenges remain aroundthe monetary valuation of outcomes to enable analysesadopting a societal perspective to be carried out morerobustly. Research addressing this issue could exploremethods that have been used before in relation to gen-eral depression [48, 49]. Similarly, if QALYs are used asoutcome, there is an urgent need to address the paucityof estimates of health state utility values relevant toPND. The limited time period inherent in trial-basedeconomic evaluations could be overcome by exploitingmodelling techniques that extrapolate outcomes andcosts over an extended timeframe [50] and the most ap-propriate time horizons could be further explored inconsultation with decision-makers. In all types of eco-nomic evaluation, robust sensitivity analyses will need tobe undertaken to explore the implications associatedwith uncertainty around outcome estimates.

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ConclusionThis systematic review has demonstrated that very feweconomic evaluations included and identified all outcomesrelevant to PND interventions. For example, outcomes forthe child were not included in most studies, and only aminority included non-health outcomes. Thus, the reviewpaves the way for further work to explore new approachesand methods that enable inclusion of relevant health andnon-health outcomes. In addition, the time horizonsadopted in the studies did not allow long-term outcomesfor the child to be addressed, which have been shown tobe important for PND. The review also shows that abroader perspective can facilitate the assessment of theoverall impact of interventions in this area. Toachieve optimal policy decisions for interventions toprevent and treat PND, addressing these methodo-logical issues is essential.

Additional files

Additional file 1: A search strategy carried out in Ovid MEDLINE (from1946 to July Week 1 2015). (DOCX 18 kb)

Additional file 2: Categorisation Criteria (details of the two-stage processused for study screening and selection). (DOCX 16 kb)

Additional file 3: Description of key methodological issues relating tooutcomes (limitations as acknowledged by the authors of the included studies).(DOCX 20 kb)

Additional file 4: Results of study assessment using Drummond’s checklist(adapted). (DOCX 21 kb)

AbbreviationsCBA: Cost-benefit analysis; CCA: Cost-consequence analysis; CEA: Cost-effectivenessanalysis; CINAHL: Cumulative Index to Nursing & Allied Health Literature; CMA: Cost-minimisation analysis; CUA: Cost-utility analysis; EPDS: Edinburgh postnataldepression scale; HTA: Health Technology Assessment; NHS EED: NationalHealth Service Economic evaluation database; NHS: National Health Service;NICE: National Institute of Health and Care Excellence; OECD: Organisationfor Economic Co-operation and Development; PND: Postnatal depression;QALY: Quality-adjusted-life-year; RCT: Randomised controlled trial

AcknowledgementsThis systematic review was undertaken at the Health Economics Unit at theUniversity of Birmingham as part of Binu Gurung’s MSc dissertation. We aregrateful to Jane Barlow (University of Warwick), Annette Bauer (The LondonSchool of Economics), Sukhdev Sembi (University of Warwick) and the membersof ‘Birmingham Perinatal Maternal and Infant Mental Health Forum’ for providing usaccess to relevant unpublished studies and data. We also thank Susan Bayliss(University of Birmingham) for her support in refining the search strategy.

Availability of data and materialsThe data on which the conclusions of the manuscript rely are presented inthe main paper and Additional files.

Authors’ contributionsLJ conceived and coordinated the study. BG assessed the title, abstract andfull texts, performed the data extraction, analysis and drafted the manuscriptwith advice from LJ, MM, RB and TR. LJ checked the coding in the two-stageprocess and assessed the full-texts. All authors read and approved the finalmanuscript.

Ethics approval and consent to participateNot applicable.

(The manuscript does not report on or involve the use of any animal orhuman data or tissue, as it is a systematic review)

Competing interestsThe authors declare that they have no competing interests.

Publisher’s NoteSpringer Nature remains neutral with regard to jurisdictional claims inpublished maps and institutional affiliations.

Author details1Health Economics Unit, Institute of Applied Health Research, College ofMedical and Dental Sciences, University of Birmingham, Edgbaston,Birmingham B15 2TT, UK. 2Cheshire and Mersey Specialist Perinatal MentalHealth Service, Thorn Road Clinic, Thorn Road, Runcorn WA7 5HQ, UK.

Received: 23 August 2016 Accepted: 10 April 2018

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