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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
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.
Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 2 of 18
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
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)
Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 4 of 18
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
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
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
Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 7 of 18
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
–
Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 8 of 18
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
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
Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 10 of 18
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
Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 11 of 18
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
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
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
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
Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 15 of 18
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.
Gurung et al. BMC Pregnancy and Childbirth (2018) 18:179 Page 16 of 18
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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