Integrated Care Models and ChildHealth: A Meta-analysisIngrid Wolfe, PhD,a Rose-Marie Satherley, PhD,a Elizabeth Scotney, MBBS,b James Newham, PhD,a Raghu Lingam, PhDc

abstractCONTEXT: Integrated care models may improve health care for children and young people (CYP)with ongoing conditions.

OBJECTIVE: To assess the effects of integrated care on child health, health service use, health carequality, school absenteeism, and costs for CYP with ongoing conditions.

DATA SOURCES: Medline, Embase, PsycINFO, Cumulative Index to Nursing and Allied HealthLiterature, and the Cochrane Library databases (1996–2018).

STUDY SELECTION: Inclusion criteria consisted of (1) randomized controlled trials, (2) evaluating anintegrated care intervention, (3) for CYP (0–18 years) with an ongoing health condition, and(4) including at least 1 health-related outcome.

DATA EXTRACTION: Descriptive data were synthesized. Data for quality of life (QoL) and emergencydepartment (ED) visits allowed meta-analyses to explore the effects of integrated carecompared to usual care.

RESULTS: Twenty-three trials were identified, describing 18 interventions. Compared with usualcare, integrated care reported greater cost savings (3/4 studies). Meta-analyses found thatintegrated care improved QoL over usual care (standard mean difference = 0.24; 95%confidence interval = 0.03–0.44; P = .02), but no significant difference was found betweengroups for ED visits (odds ratio = 0.88; 95% confidence interval = 0.57–1.37; P = .57).

LIMITATIONS: Included studies had variable quality of intervention, trial design, and reporting.Randomized controlled trials only were included, but valuable data from other study designsmay exist.

CONCLUSIONS: Integrated care for CYP with ongoing conditions may deliver improved QoL andcost savings. The effects of integrated care on outcomes including ED visits is unclear.

aDepartment of Women’s and Children’s Health, School of Life Course Sciences, Faculty of Life Sciences and Medicine, King’s College London, London, United Kingdom; bKing’s College HospitalNational Health Services Foundation Trust, London, United Kingdom; and cPopulation Child Health Clinical Research Group, School of Women and Children’s Health, University of New SouthWales, Sydney, New South Wales, Australia

Dr Wolfe conceptualized and designed the study, supervised data collection and analysis, and is accountable for all aspects of the work; Dr Lingam conceptualized anddesigned the study and supervised data collection and analysis; Dr Satherley contributed to conceptualizing and designing the study, wrote the first draft, designed thedata collection instruments, collected data, and conducted the initial analyses; Dr Scotney contributed to conceptualizing and designing the study and collected data; DrNewham completed the meta-analysis; and all authors reviewed and revised the manuscript and approved the final manuscript as submitted.

DOI: https://doi.org/10.1542/peds.2018-3747

Accepted for publication Oct 4, 2019

Address correspondence to Ingrid Wolfe, PhD, Department of Women’s and Children’s Health, School of Life Course Sciences, Faculty of Life Sciences and Medicine,King’s College London, 5th Floor Becket House, 1 Lambeth Palace Rd, London SE1 7EU, United Kingdom. E-mail: Ingrid.wolfe@kcl.ac.uk

PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).

Copyright © 2020 by the American Academy of Pediatrics

To cite: Wolfe I, Satherley R, Scotney E, et al. Integrated Care Models and Child Health: A Meta-analysis. Pediatrics. 2020;145(1):e20183747

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Child mortality has decreasedmarkedly over the last 50 years, andthe epidemiological transition towardchronic conditions, such as asthmaand neurodevelopmental disorders,applies across many countries andregions.1–5 These broad population-level changes mean that healthsystems and health care models mustadapt accordingly, providing high-quality care for all childhoodconditions.

International data indicate thatcurrent models of care are inadequateand unsustainable. In England, therewas a 58% rise in children and youngpeople (CYP) attending hospitalemergency departments (EDs) from2007 to 2016,6 with a further 50% to60% increase projected by 2030.7

Eighty-five percent of current EDattendances by CYP are of lowseverity,8 manageable in primarycare, and the number of childrenpresenting to EDs with minorailments increases 5% annually.9 Inthe United States, only 47% of clinicalcontacts adhere to clinical guidelinesacross 11 conditions (2007)10 and60% across 17 conditions in Australia(2018).11

Authors and policy makers haveargued that the current health caremodel, in the context of the widerhealth and social system, deliverssuboptimal health outcomes forchildren.12–15 However, new modelsof integrated care are largely focusedon adults, with adults’ needs in mind,including multiple comorbidities andfrailty.16 Definitions of integrated carevary, but consistent themes that applyto any population include person-centered care delivered byprofessionals and organizationscooperating effectively. Integratedcare is thought to increase the patientfocus of care and deliver more cost-effective health care.17 For adults,integrated care has been associatedwith positive outcomes, with someevidence for reductions in hospitalactivity and costs.18–24 For children,there are reviews and meta-analyses

examining specific conditions orcircumstances, for example byAsarnow et al25; however, we foundno systematic reviews examiningevidence for comprehensive broadpopulation-based integrated care forCYP. In this systematic review, weassess the impact of integrated careinterventions for CYP with ongoingconditions on important outcomes forchildren including health, healthservice use, health care quality,education, and costs.

METHODS

We conducted a systematic literaturesearch (1990–2018) following thePreferred Reporting Items forSystematic Reviews and Meta-Analyses guidelines. The protocol forthis review was registered in theInternational Prospective Register ofSystematic Reviews(CRD42016045215).

Two authors completed the search inJuly 2017, which was updated inSeptember 2018. Five databases weresearched: Medline, Embase,PsycINFO, Cumulative Index toNursing and Allied Health Literature,and the Cochrane Library. Twoauthors independently screenedtitles, abstracts, and full texts againstthe inclusion criteria. A third authorinformed final decisions whendisagreements occurred.

Search criteria comprised 3categories: (1) population (age,condition), (2) intervention(integrated care), and (3) studydesign (see SupplementalInformation for the full searchstrategy). Additional articles wereidentified through reference trackingof included studies. Studies wereeligible for review if they (1) reportedrandomized controlled trials (RCTs)published between 1990 andSeptember 2018, (2) evaluated anintegrated health care interventiondesigned to improve CYP’s health, (3)included CYP (0–18 years) with anongoing health condition (defined as

any health problem requiring clinicalfollow-up for .12 months in 50% ormore cases),26 (4) included at least1 health-related outcome indicator,and (5) were a peer-reviewedpublication available in English.Eligible articles were screened toidentify references reportingadditional results from the describedintervention, which were included ifthey assessed the same interventionand sample as the original article.

Integrated Care Definition

Many definitions of integrated carehave been used in practice and thescientific literature, but nostandardized widely accepteddefinition exists.27 The World HealthOrganization defines integrated careas “health services organized andmanaged so that people get the carethey need, when they need it, in waysthat are user-friendly, achieve thedesired results and provide value formoney.”28 This broad definitiondescribes only the principles ofintegrated care and is not suitable toassess the presence or extent ofintegration. For practical applicationand to reflect the different integrationneeds of CYP contrasted with adults,we use the Wolfe et al15 definition,which adapts the World HealthOrganization principles of integrationinto domains specifically for theindividual- and population-levelhealth care needs of children. Thefirst domain is vertical integrationbetween primary and secondary care,which is important at the populationlevel because minor and acute illnessmanagement is a large part ofchildren’s health service use and iscrucial for effective gatekeeping. Anexample is general practitioners orprimary care pediatricians workingeffectively with secondary carepediatricians and mental healthprofessionals. Horizontal integrationis both between the health andeducation sectors, which is importantfor all CYP, and integration betweenhealth and social care, which isimportant for CYP with complex

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needs. Finally, longitudinalintegration is important for age-appropriate care, at key times ofdevelopmental transition (eg,between pediatric and adult services).We used these categories ofintegration to identify if anintervention was integrated andwhich domain applied. Interventionsthat included $1 of these integrationtypes were eligible for inclusion.

Data Extraction and QualityAssessment

Interventions were defined as“integrated care” (yes or no) for eachof the domains of integrationdescribed above,15 with a detailedrationale documented explaining howthe intervention met these criteria.Data were extracted from reports ofinterventions integrated in at least 1domain, with outcomes relating to (1)child health outcomes, (2) healthservice use, (3) health care quality,(4) education (school absenteeism),and (5) cost savings and cost-effectiveness. Population dataincluding sociodemographic details,comparator groups, intervention typeand category of integration, studydesign, and outcome measures wereextracted. Bias was assessed by usingthe Cochrane Collaboration tool.29

Two authors assessed the articles andextracted data, with discrepanciesresolved by discussion. Whenprotocol articles were available, thesewere consulted to assist in dataextraction and quality assessment.

Data Analysis

Meta-analyses of the effectiveness ofinterventions were conducted on datafrom studies in which there washomogeneity of the outcome variable:rates of ED visits and child-relatedquality of life (QoL) measures. Forthese 2 outcome measures, whenpossible, we summarized data at3 follow-up time points (3, 6, and 12months) and when reported, datafrom intention-to-treat analyses wereused. If 2 interventions werecompared against a control group,

data from both intervention armswere included, and the number ofparticipants in the control group washalved for each comparison.Postintervention outcomes, reportedas mean and SD, were used foranalysis; when postinterventionscores were unavailable, mean changescores were used. When mean and SDvalues were unavailable, missing datawere imputed by estimating the SDfrom the confidence intervals (CIs).Standardized mean differences(SMDs) with 95% CIs were calculatedand pooled by using a random effectsmodel for all studies. Dichotomousand continuous outcomes weremerged by using ComprehensiveMeta-Analysis software (version 2.2;Biostat, Englewood, NJ) to produceSMDs (equivalent to Cohen’s d) foreach study. SMD values of at least 0.2,0.5, and 0.8 indicate small, medium,and large effect sizes, respectively.30

Heterogeneity across studies wasassessed by using Cochran Q test andI2 test statistics.

Integration was defined as present orabsent (as specified above), andbecause there is no establishedmeasure of extent of integration,a description of the integrated servicewas obtained. Because we includeda wide breadth of interventions tocapture the diversity of interventionsfor any and all children’s health care,we used descriptive assessment andclinical judgment of included articlesto support the statistical assessmentof homogeneity.

RESULTS

The search generated 3956 articles;23 articles, reporting 18interventions, met the inclusioncriteria (Fig 1). Risk of bias overallwas moderate; a lack of blindingparticipants was the most commonreason for downgrading quality (n = 8interventions). An overview of resultsis provided in Table 1 andSupplemental Table 2.

Study Characteristics

Sample sizes ranged between 24 and1316 CYP, and the mean age was 8.9years. Asthma was the mostcommonly studied condition (n = 6).Most (n = 13) studies were conductedin North America.31–44 Comparisongroups were treatment as usual (n =14), waitlist control (n = 2), orenhanced usual care (n = 2); however,a description of usual care wastypically not provided.

The 18 interventions included in thisreview are described in Table 1.Gorelick et al38 and Seid et al44

compared 2 integrated interventionsto treatment as usual. Whenoutcomes for the same interventionand patient sample were reportedacross several articles,34–36,40,41,45–48

the results were combined to reflectthe overall impact of the intervention.

Intervention Characteristics

All interventions were focused onimproving child health; however, thecontent differed. All articles describedthe evidence used for interventiondevelopment; however, there waslimited detail on hypothesizedmechanisms of action provided, andno article included a logic model.Most interventions were complex,incorporating multiple elements toachieve integration, summarized inTable 1. Ten interventions includedaspects of verticalintegration,31,38,40,41,43–50 10 includedhorizontalintegration,32–37,39,40,42,44,51 and nointerventions described longitudinalintegration.

Study Outcomes

Results are described according tothe 5 categories defined in the aims ofthis review: (1) child healthoutcomes, (2) health service use, (3)health care quality, (4) education(school absenteeism), and (5) costsavings and cost-effectiveness. Meta-analysis was possible only for childhealth outcomes (QoL) and health

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service use (ED visits) because ofdata heterogeneity.

Child Health Outcomes (18Interventions)

Health Status and Functional Symptoms

Studies of 14 interventions assessedchanges in 19 functional healthindicators, such as improvement incondition-related symptoms. Thequality of evidence was moderate(42.9% at low risk of bias). Five ofthese 19 indicators (26.3%) revealedan improvement among CYP in theintervention group, compared withcontrols.34–36,39–41,44,52 Theseindicators included hemoglobin A1c(a measure of diabetes control), pain,disability, symptoms of attention-deficit/hyperactivity disorder, viralload, and symptoms of asthma. Effectsizes for comparisons acrossintervention and control were notreported. Authors of only 1 articleassessed maintenance of effect,

finding improved functional status forthe intervention compared to thecontrol at 12 months but notmaintained at 18 months.49

All interventions in which authorsreported a significant healthimprovement effect for integratedcare, compared to the control,included education for caregivers.Regarding improvements in asthmasymptoms, Seid et al44 compared 2integrated interventions to controls(rated at low risk of bias), whichrevealed contrasting results 3 monthspostintervention. Both integratedcare interventions reported aspects ofvertical and horizontal integration:hospital service providerscommunicated with primary careproviders via the electronic healthrecord and referred to communityservices. CYP receiving integratedcare interventions had a one-thirdodds of having .1 asthma-relatednighttime symptom per week

compared with controls but onlywhen a psychoeducational workshopfor parents was included.

QoL

Studies of 8 interventions assessedchanges in QoL between interventionand controls; the quality of evidencewas good (62.5% at low risk of bias).Authors of 3 interventions reportedgreater improvements in QoL for theintervention compared to the control.A meta-analysis to quantify the size ofeffect for QoL was only possible withdata for 6-month follow-up. Meta-analyses (see Fig 2) revealed thatinterventions had a significantpositive effect in improving QoLscores over usual care in 4 studies (5comparisons) when data wereavailable for pooling (SMD = 0.24;95% CI = 0.03–0.44; P = .02). Therewas no statistical heterogeneitybetween studies (I2 = 0%; P = .48);however, studies were heterogeneousregarding clinical populations andcontrol group characteristics. Authorsof 1 article compared the interventionto enhanced usual care31; the authorsof the remaining articles comparedthe integrated health careintervention to treatment as usual.The condition studied varied acrossarticles and included diabetes,45–48

asthma,44 and obesity.31,50

Psychological Indicators

Studies of 8 interventions assessedchanges in 14 psychologicalindicators across the intervention andcontrol; the quality of evidenceoverall was poor (2 at low risk ofbias). Indicators included anxiety (n =2), distress and depression (n = 4),psychosocial functioning and/oradjustment (n = 2), well-being (n = 1),body dissatisfaction (n = 1),behavioral symptoms (n = 1), andself-esteem or self-worth (n = 3). Ofthese 14 indicators, statisticallysignificant improvements were foundfor 5 of 14 indicators (35.7%) whencompared with controls.Psychological outcomes varied withinstudies. For example, Glisson et al37

FIGURE 1Preferred Reporting Items for Systematic Reviews and Meta-Analyses diagram of search results. Atotal of 299 articles did not meet the inclusion criteria at full-text screening regarding intervention(n = 142), population (n = 87), study design (n = 45), comparator (n = 7), full text not available (n =6), duplicate (n = 2), outcome (n = 10).

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found no effect of integrated careinterventions on well-being, whichimproved in both intervention andcontrol groups during the study period,but girls receiving integrated careshowed significant improvements inself-efficacy (effect sizes acrosssubscales range between 0.41 and0.69) compared with girls receivingusual care who showed a significantdecline in self-efficacy. No changes inself-efficacy were found for boys acrossthe control and intervention arms.

Health Service Use (n 5 10Interventions)

Studies of 9 interventions reportedhealth service use; the quality ofevidence overall was moderate (20%at low risk of bias).

ED Visits

Studies of 7 interventions assessedED visits.33–36,38,44,49 In all studies,authors hypothesized that integratedcare interventions would reduce EDvisits. There was sufficient evidenceto examine the effect of interventionson visits to the ED (measured byparent recall) with 2 comparisons(from 2 studies) but only at 6-monthfollow-up. Meta-analyses (Fig 3)revealed that integrated care did notsignificantly reduce ED visits betweeninterventions and control groups(odds ratio [OR] = 0.88; 95% CI =0.57–1.37; P = .57). There was nostatistical heterogeneity betweenstudies (I2 = 0%; P = .74).

Primary and Secondary Care Use

Six studies used a variety ofindicators to assess the impact onsecondary care use. These includedattendance at mental health services(n = 1), urgent doctor appointments(n = 2), inpatient visits (n = 4), days inhospital (n = 2), and length of stay(n = 1); no studies examined primarycare use. The hypothesized outcomeassociated with integrated careinterventions varied across articles,with some aiming to increase CYPcontact with the health care serviceand others to reduce contact. Overall, TA

BLE1Characteristicsof

Interventions

Included

inReview

Article

Bankset

al31

Chernoffet

al32

Cicuttoet

al33

Ellis

etal34–36

Glissonet

al37

Gorelicket

al38

Gorelicket

al38

Grupp-Phelan

etal51

Hechleret

al39

Condition

Obesity

Chronicillness

Asthma

Diabetes

Mentalhealth

Asthma

Asthma

Suiciderisk

Chronicpain

Outcom

eChild

health

Health

status

↔—

—↑

—↔

↔—

↑QoL

↔—

↑↑

——

——

—

Psychological

—↑↔

—↑

↑—

—↔

↔Health

service

use

EDuse

——

↑↑

—↔

↔—

—

1ry,2rycare

use

——

—↑

——

—↔↑

—

Health

care

quality

↔—

——

——

—↔

—

Education

——

↑—

——

——

↔Cost

savings

——

—↑

——

——

↑Integrated

intervention;

setting;

summary

results;risk

ofbias

Vertical;1ry

care

nurses

and2ry

care

exercise

consultant

and

dietician;England;

intervention

andcontrol

equally

effective

aseach

othera

Horizontal;child

health

social

services;

UnitedStates;

positive

effectsof

intervention

forpsychological

functioning

b

Horizontal;

publichealth

nurses

and

school

staff;

UnitedStates;

positive

effectsof

interventionb

Horizontal;

therapists

andschool

staff;United

States;positive

effectsfor

intervention,

maintained

at24

mob

Horizontal;

health

and

education

sectors;

UnitedStates;

positive

effectsof

interventionc

Vertical;1ry

care

and

2rycare

asthmateam

;UnitedStates;

intervention

andcontrol

wereas

effectiveas

each

otherc

Vertical;1ry

care

and2rycare

asthma

team

,horizontalhealth

andsocial

services;

UnitedStates;

interventionand

controlwereas

effectiveas

each

otherc

Horizontal;m

ental

health

andsocial

worker

coordinating

care;U

nited

States;positive

effectsfor

interventionfor

health

service

usec

Horizontal;

health

and

education,care

bya

multidisciplinary

team

;Germany;

positiveeffects

forinterventionc

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TABLE1Continued.

Characteristicsof

Interventions

Included

inReview

Article

Kolkoet

al40;

Yuet

al41

Letourneau

etal52

McW

hirter

etal42

O’Shea

etal43

Seid

etal44

Seid

etal44

Simon

etal49

Tilberget

al45–48

Wakeet

al50

Condition

Behavioral

problems

HIV

Asthma

Chroniclung

disease

Asthma

Asthma

Medical

complexity

Diabetes

Obesity

Outcom

eChild

health

Health

status

↑↔

↑↔

↔↔

↑↔

↔↔

QoL

↑—

↔—

↔↑

—↔

↔Psychological

——

↔↑

↔—

——

—↔

Health

serviceuse

EDuse

——

——

↔↔

↔—

—

1ry,2ry

care

use

——

——

↔↔

↑↑

—

Health

care

quality

↑↔

——

——

↑↔

↑—

Education

——

↔—

——

——

—

Cost

savings

↑—

——

——

↓↑↔

—

Integrated

intervention;

setting;

summary

results;

risk

ofbias

Vertical;1ry

care

andbehavioral

health

services;

UnitedStates;

positiveeffects

ofintervention

notmaintained

at18-mo

follow-upa

Horizontal;

health

andsocial

services;

UnitedStates;

intervention

andcontrol

wereas

effective

aseach

otherc

Horizontal;

nurseand

school

staff;

England;

positive

effectsfor

intervention

forchild

health

(viral

load)c

Vertical

1rycare

and2rycare

nurseand

neonatologist;

UnitedStates;

interventionand

controlas

effective

aseach

othera

Vertical;hom

ehealth

visitors

and1rycare,

horizontal;

community

care

services,

care

coordination;

UnitedStates;

intervention

andcontrolas

effectiveas

each

othera

Vertical;hom

ehealth

visitors

and1rycare,

horizontal;

community

care

services,care

coordinationwith

psychoeducational

workshop;

United

States;p

ositive

effectsfor

interventionover

controlfor

child

health

a

Vertical;

multidisciplinary

team

in2ry

care

and1ry

care

provider;

UnitedStates;

positiveeffects

ofintervention

forquality

ofcare

notmaintained

at18

moand

serviceuse;

intervention

moreexpensivea

Vertical

integration

between

health

(diabetes

nurse)

andschool

staff;Sw

eden;

positiveeffects

forintervention

forhealth

serviceuse,

quality

ofcare,and

costsc

Vertical

integration

between

pediatrician

anddieticianin

2rycare,and

generalpractitioner;

Australia;

intervention

andcontrolwere

aseffectiveas

each

othera

1ry,primary;2ry,secondary;↑,

outcom

ereported

statisticallysignificant

change

infavorof

intervention;

↓,in

favorof

control;↔,nosignificant

differences

betweeninterventionandcontrol;—

,datanotavailable.

aForrisk

ofbias,green

indicateslowrisk.

bForrisk

ofbias,red

indicateshigh

risk.

cForrisk

ofbias,amberindicatesmoderaterisk.

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4 of 10 indicators met their desiredoutcome and revealed greaterimprovements in secondary care usefor integrated care over control.

Simon et al49 reported that the use ofa multidisciplinary team and sharedcare records between the secondaryand primary care provider resulted ina greater reduction in total hospitaldays and length of stay (.7 days) forCYP with medical complexity whencompared to treatment as usual overa period of 12 months. Improvementswere maintained at 18 monthspostintervention for length of stay(.7 days) but not for total hospitaldays. However, no change was foundbetween the intervention and controlfor inpatient admissions, length ofstay (per child-year), or ICU visits. Noeffect sizes were reported, but the

authors anticipate that theintervention has increasedaccessibility to health care servicesthrough its focused care coordination.Grupp-Phelan et al51 used anintegrated intervention aiming toenhance access and increase contactwith mental health services. The trialreported no difference among CYPreceiving the intervention compared tothe control regarding schedulinga mental health appointment (OR =2.29; 95% CI = 0.41–12.7/1.0) butgreater likelihood of attending theirfirst mental health appointment at60 days postintervention (OR = 9.62;95% CI = 1.38–67.25/1.0).

Health Care Quality (n 5 6Interventions)

Six studies reported indicators forhealth care quality; 5 of these were

related to service satisfaction. Thequality of evidence among includedstudies was moderate. One article49

included a combined measure ofseveral indicators of health carequality including support forcoordinating multiple providers,getting care needed quickly,prescription medication, gettingspecialized services, shared decision-making, and getting neededinformation. Authors of 2 studiesreported increased satisfaction withintegrated interventions whencompared with controls40,41,45–48;however, no further information toquantify these effects was provided.For the combined measure of healthcare quality, Simon et al49 reportedgreater improvements for theintervention over the control at12 months postintervention, but

FIGURE 2Forest plot of effect size of association between intervention and QoL at 6-month follow-up. Heterogeneity: I2 = 0%; P = .48. CC, care coordination andproblem-solving intervention; CC with PST, care coordination and problem-solving intervention with psychoeducational workshop.

FIGURE 3Forest plot of effect size of association between intervention and ED at 6-month follow-up. Heterogeneity: I2 = 0%; P = .74. CC, care coordination andproblem-solving intervention; CC with PST, care coordination and problem-solving intervention with psychoeducational workshop; PCL, primary carelinkage.

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these effects were not maintained at18 months. For individual indicators,there was a greater impact. Forexample, help coordinating multipleproviders and getting care quicklywere improved for the intervention,over the control, and were sustainedat 18 and 12 months, respectively.

Education (n 5 3 interventions)

Three studies were used to assessschool absenteeism as an education-related outcome regarding integratedhealth care. Authors of 1 study foundstatistically significant reductions inschool absenteeism for theintervention compared to thecontrol,33 reporting a 69% reductionfor school absenteeism among CYPwith asthma; no estimates of effectsize were reported.

Cost Savings and Cost-effectiveness(n 5 5 Interventions)

Four studies included a cost-savingsanalysis of integrated health careinterventions compared tocontrols34–36,39–41,49; authors of 1study reported analysis of cost-effectiveness.45–48 The quality ofstudies overall was moderate (2 atlow risk of bias). All studies assessedchanges in health care costs from thehealth system perspective, usinghealth service use data, and 2 alsoassessed the societalperspective.39,45–48 Overall costsavings were reported for 3 of 4interventions when compared to thecontrol; no difference was foundbetween the intervention and controlfor cost-effectiveness.45–48

From the health system perspective,integrated care interventions weremore expensive to implement whencompared to controls,34–36,39–41,49 butthese costs were offset by savingsfrom health care usage.34–36,40,41 Forexample, Yu et al41 reported a lowercost per integrated care patientassociated with mental healthservices, when compared withcontrols, during the 6-month trial andat 6- to 12-month follow-up points.

However, these differences narrowedat 18 to 24 months as CYP movedfurther from the intervention period.For Simon et al,49 the highimplementation costs were not offsetby cost savings over an 18-monthperiod.

From the societal perspective,Hechler et al39 provided CYP andfamilies with self-managementsupport and worked alongsideschools. This intervention wasassociated with significant reductionsin parental financial burdencompared with controls, and indirectcosts to employers decreased asparental work absenteeism decreasedsignificantly. Ellis45–48 found nochanges in cost savings from thesocietal perspective.

DISCUSSION

This is the first article to presenta systematic assessment of theeffectiveness of integrated health careinterventions on important outcomesfor children with ongoing conditionsincluding health, health service use,health care quality, education, andcost savings. Using a broad systematicsearch, we identified varied, and oftencontradictory, outcomes among 18integrated care interventions. Studieswere often limited by poor reportingof statistics or the quality of evidence.

Overall, integrated care for CYP withongoing conditions appears to offerstatistically significant improvementsin health-related QoL. However, forother child health outcomes, healthservice use, health care quality,education (school absenteeism), andcost savings and cost-effectiveness,the evidence is mixed.

Caregiver and education involvementappear to be important in deliveringpositive changes in CYP health status.At the health system level, theevidence about the influence ofintegrated care on secondary careusage was mixed and depended onwhether the intervention aimed toincrease accessibility of services or

reduce reliance on secondary careservices. Surprisingly, no articlesreported on the impact of integratedcare on primary care usage.

The meta-analysis indicated limitedeffectiveness of integrated care inreducing ED visits for CYP withongoing conditions. However, thebroad CIs highlight the heterogeneityof the evidence on the impact ofintegrated care on ED visits. Of note,the integrated care interventionsdescribed in this review did nottarget minor ailments that cause largenumbers of ED visits in CYP withongoing conditions. Our findings onED attendance contrast with recentobservational studies that haveshown up to a 40% decrease in EDpresentations for children withmedical complexity.53

This review highlights importantconcerns about the design andevaluation of children’s integratedcare. Information regarding thedesign of integrated interventions inthe articles included in this review islimited because no studies presentedor described the use of logic modelsto inform complex interventiondevelopment, which have been shownto improve interventionefficiency54,55 or the use ofappropriate theoretical frameworksto guide the selection of interventionsand outcome indicators. Perhaps asa result of these omissions, theinterventions in this review werechallenging to describe because of thecomplex nature of overlappingintervention components.

Evaluation of integrated careinterventions among the articlesincluded was limited by variablequality (despite selecting only RCTs),heterogeneity of outcomes and lack ofstandardized outcome measures, andinsufficient follow-up periods. Thecontrasting results between studiesusing observational and trial designsand the variable quality of publishedstudies suggests there is much to begained from further high-quality

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research that is focused on integratedhealth care models for CYP.Additionally, although heterogeneityprevented the comparison of studiesassessing health care costs in thisreview, authors of 3 of 4 articlesreported cost savings associated withintegrated care despite higherimplementation costs. However, it isnot clear whether these cost savingswere sustained. Again, standardizedmeasures and appropriate follow-upperiods are essential to improve thequality of evidence available.

Recommendations

With this review, we presenta synthesis of the best availabledata on integrated care for CYP. Wefind that integrated care may deliverimprovements in QoL and may offercost savings; however, there isinsufficiently strong evidence currentlyto make firm recommendationsregarding the overall effectiveness ofintegrated services.

Our findings highlight important gapsin the evidence. We recommend thatfuture studies should followestablished guidelines on thedevelopment and evaluation ofcomplex health systeminterventions.56 Using logic modelsand intervention design methods thatinclude behavior change mechanismsat the level of the individual and thesystem are also important.57 Agreeddefinitions of integration anda validated measure to assess thelevel of integration in health careinterventions would be useful.

To improve the quality and usefulnessof research, robust evaluation design,standardization of outcome measures(eg, ED visits), and appropriate follow-up periods are essential, as isinformative standardized reporting, forexample, of effect sizes betweenintervention and control groups.

Strengths and Limitations

This is the first systematic review ofchildren’s integrated careinterventions that includes

a comprehensive variety ofinterventions to capture the diversityneeded for all children’s health care.However, the breadth of interventionsincluded also brought challenges.Articles were limited in theirdescriptions of usual care, makingcomparison with control groupschallenging. In addition, with theavailable data, we could not discernwhich children benefited most fromintegrated health care. Severity ofsymptoms and socioeconomicbackground may be importantdeterminants that influence health careoutcomes. Furthermore, no conclusionscould be made about the extent ofintegration that may be beneficial forchild health or system measuresbecause there is currently no validatedmeasure of extent of integration.

Of the 18 interventions identified,studies on 6 interventions reported onCYP with asthma. Eleven of 18 studieswere set in North America; therefore,generalizability of the findings may belimited to these groups, particularlygiven the differing nature of health caresystems.

Despite low statistical heterogeneity,descriptive analysis indicated thatstudies included in the meta-analysesdiffered in interventioncharacteristics and evaluation design;therefore, the meta-analysis needs tobe interpreted with a clearunderstanding of these inherentchallenges of health servicesresearch.58 To aid interpretation inthe context of these contrastingstatistical and descriptive findings,we have presented detaileddescriptions of each study included inour study tables and highlighted theimpact of each study in turn. This isintended to support the reader toassess the impact of integrated carein each outcome domain(Supplemental Table 2).

Implications for Clinicians and PolicyMakers

The evidence for integrated careinterventions improving child health

is limited but encouraging. However,the best available data are currentlyinsufficiently robust to make firmrecommendations regarding theoverall effectiveness of integratedservices to improve children’s health.We recommend strongly thatresearch efforts are directed urgentlytoward improving the design andevaluation of integrated health caremodels for children.

CONCLUSIONS

Health systems need to adapt toimprove quality and meet evolvinghealth needs, so there is an urgentneed for high-quality evidence.Integrated models of care may deliversome important improvements inQoL for CYP with ongoing healthconditions and potentially savemoney for the stretched healthsystem. However, the evidence baseremains insufficient, and there is anurgent need for high-quality researchto develop and evaluate theoreticallyframed integrated models of care tohelp CYP living with ongoing healthconditions.

ACKNOWLEDGMENTS

We thank Julia Forman of theDepartment of Women’s andChildren’s Health, School of LifeCourse Sciences, Faculty of LifeSciences and Medicine, King’sCollege London, and MohamedElsherbiny of the Department ofPopulation Health Sciences, King’sCollege London.

ABBREVIATIONS

CI: confidence intervalCYP: children and young peopleED: emergency departmentOR: odds ratioQoL: quality of lifeRCT: randomized controlled trialSMD: standardized mean

difference

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FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.

FUNDING: No external funding.

POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose.

COMPANION PAPER: A companion to this article can be found online at www.pediatrics.org/cgi/doi/10.1542/peds.2019-3282.

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