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SYSTEMATIC REVIEW
Social determinants of health and outcomes for children and
adults with congenital heart disease: a systematic review
Brooke Davey
1,2
, Raina Sinha
2,3
, Ji Hyun Lee
2,4
, Marissa Gauthier
5
and Glenn Flores
2,6
BACKGROUND: Social determinants of health (SDH) can substantially impact health outcomes. A systematic review, however, has
never been conducted on associations of SDH with congenital heart disease (CHD) outcomes. The aim, therefore, was to conduct
such a systematic review.
METHODS: Seven databases were searched through May 2020 to identify articles on SDH associations with CHD. SDH examined
included poverty, uninsurance, housing instability, parental educational attainment, immigration status, food insecurity, and
transportation barriers. Studies were independently selected and coded by two researchers based on the PICO statement.
RESULTS: The search generated 3992 citations; 88 were included in the final database. SDH were significantly associated with a
lower likelihood of fetal CHD diagnosis, higher CHD incidence and prevalence, increased infant mortality, adverse post-surgical
outcomes (including hospital readmission and death), decreased healthcare access (including missed appointments, no shows, and
loss to follow-up), impaired neurodevelopmental outcomes (including IQ and school performance) and quality of life, and adverse
outcomes for adults with CHD (including endocarditis, hospitalization, and death).
CONCLUSIONS: SDH are associated with a wide range of adverse outcomes for fetuses, children, and adults with CHD. SDH
screening and referral to appropriate services has the potential to improve outcomes for CHD patients across the lifespan.
Pediatric Research _#####################_ ; https://doi.org/10.1038/s41390-020-01196-6
IMPACT:
●Social determinants of health (SDH) are associated with a wide range of adverse outcomes for fetuses, children, and adults with
congenital heart disease (CHD).
●This is the first systematic review (to our knowledge) on associations of SDH with congenital heart disease CHD outcomes.
●SDH screening and referral to appropriate services has the potential to improve outcomes for CHD patients across the lifespan.
BACKGROUND
Innovation and technical advancement have revolutionized the
field of pediatric and adult congenital heart disease (CHD) over
the past century. As clinical outcomes have improved drama-
tically over time, however, healthcare disparities have persisted
for the most vulnerable populations. Structural cardiac defects
are the most common birth defect, affecting approximately
0.8–1% of the population.
1–3
These birth defects range in
complexity and occur across all the socioeconomic groups. With
CHD mortality in infancy and childhood decreasing substan-
tially with the evolution of advanced surgical and catheter-
based interventions, >90% of children with CHD now survive
into adulthood, and this large population of adults with CHD
continues to grow with time.
4
As a result, there are now more
adults than children living with CHD in the US. Although
survival has improved, CHD patients continue to face major
socioeconomic and demographic disparities in outcomes at
all ages.
3
Social determinants of health (SDH) are conditions in which
people live and grow up within the wider context of systems and
influences shaping daily life.
5
SDH include poverty, lack of insurance,
housing instability, parental educational attainment, immigration
status, food insecurity, and transportation barriers. These factors
contribute to poor clinical outcomes, healthcare inequities, and
escalating healthcare costs. The central importance of the associa-
tion of SDH with health outcomes specifically in the context of
cardiovascular diseases was underscored by the American Heart
Association and American College of Cardiology in their 2019
guidelines for clinical risk assessment.
6
There are no published
systematic reviews, however, of the associations of SDH with major
CHD outcomes across the lifespan, including fetal diagnosis;
incidence and prevalence; infant mortality; post-surgical outcomes;
access to care, loss to follow-up, and hospital readmissions;
neurodevelopmental outcomes and quality of life (QOL); and adult
CHD. The study aim, therefore, was to conduct a systematic review
of the association of SDH with CHD outcomes.
Received: 7 July 2020 Revised: 2 September 2020 Accepted: 10 September 2020
1
Division of Cardiology, Connecticut Children’s Medical Center, Hartford, CT, USA;
2
Department of Pediatrics, University of Connecticut School of Medicine, Farmington, CT, USA;
3
Division of Cardiac Surgery, Connecticut Children’s Medical Center, Hartford, CT, USA;
4
Research Department, Connecticut Children’s Medical Center, Hartford, CT, USA;
5
Health
Sciences Library, University of Connecticut School of Medicine, Farmington, CT, USA and
6
Health Services Research Institute, Connecticut Children’s Medical Center, Hartford, CT,
USA
Correspondence: Glenn Flores (gflores@connecticutchildrens.org)
www.nature.com/pr
©International Pediatric Research Foundation, Inc 2020
1234567890();,:
METHODS
SDH: definitions
The following SDH were included in this analysis: poverty,
uninsurance, housing instability, parental educational attainment,
immigration status, food insecurity, and transportation barriers.
These were chosen because they are the domains addressed in a
recently published SDH screening instrument used for interven-
tions effective in reducing social risks and improving child and
caregiver health.
7
For articles in which there was no assessment of
socioeconomic status (SES), Medicaid coverage was used as a
proxy for low income.
Outcomes
The following CHD outcome categories across the lifespan were
evaluated: fetal diagnosis; incidence and prevalence; infant
mortality; post-surgical outcomes; access to care, loss to follow-
up, and hospital readmissions; neurodevelopmental outcomes
and QOL; and adult CHD.
Inclusion and exclusion criteria
Inclusion criteria consisted of published, original research on
the associations of SDH with CHD. Exclusion criteria included:
(1) letters to the editor, commentaries, editorials, viewpoints,
perspectives, opinion pieces, case reports, book chapters, author
or keyword indexes, and review articles; (2) publications that did
not address clinical outcomes in patients with CHD; (3) articles
focusing on acquired pediatric cardiac diseases, variants of
normal, patent foramen ovale, primary arrhythmias, cardiovascular
complications of connective tissue disorders, and pulmonary
hypertension in the absence of structural CHD; (4) studies that
classified SDH as race/ethnicity, maternal stress, environmental
exposures, or vitamin or drug/alcohol exposures; (5) animal-only
studies; (6) analyses of the association of CHD (as an independent
variable) with SDH (as the dependent variable); and (7) articles on
populations outside of the US or Canada (because the focus was
on SDH in developed countries in North America with comparable
healthcare systems).
Literature search
Using sentinel articles to harvest and test search terms, the
following search strategy was developed for PubMed/MEDLINE to
retrieve all records using natural language and controlled
vocabulary (when available) relating to the association of SDH
with CHD (Table 1). This strategy then was translated and adapted
for the other databases. The following databases were searched
from date of inception through May 18, 2020: PubMed MEDLINE
(including Pre-MEDLINE and non-MEDLINE; 1945 to May 2020),
Scopus (Elsevier; 1966 to May 2020), Cochrane Central Register of
Controlled Trials (Wiley; through May 2020), CINAHL (Ebsco; 1981
to May 2020), PsycInfo (Ebsco; 1872 to May 2020), Social
Interventions Research & Evaluation Network (SIREN) Evidence &
Resource Library (University of California, San Francisco; through
May 2020), and SocIndex (Ebsco; 1895 to May 2020). No filters
were used for language or publication date. ProQuest RefWorks
(Legacy version) was used to de-duplicate and manage all
citations.
Once articles were identifiedandcompiledbythesearch
criteria described above and duplicates removed, vetting was
performed by title and abstract by two authors using strict
inclusion and exclusion criteria (Fig. 1).Forstudiesforwhich
there was lack of clarity regarding whether or not inclusion
criteria were met, final decisions were made by reaching
consensus among at least three authors. Once title and abstract
vetting was completed, a full-text review was performed
using the inclusion and exclusion criteria to determine final
inclusion in the systematic review. Consensus opinion with
regards to inclusion of studies was again reached when
questions arose.
PROSPERO registration
This systematic review was registered in PROSPERO
(CRD42020169253).
RESULTS
The initial search generated 3992 citations. A total of 88 studies
met inclusion criteria (Fig. 1and Table 2). Study designs were
variable and included retrospective chart reviews, retrospective
and prospective cohort studies, cross-sectional studies, and
prospective case–control studies. No studies were identified that
examine housing instability.
The sections that follow report the findings on SDH associations
with seven major CHD outcomes across the lifespan: fetal
diagnosis; incidence and prevalence; infant mortality; post-
surgical outcomes; access to care, loss to follow-up, and hospital
readmissions; neurodevelopmental outcomes and QOL; and
adult CHD.
Fetal diagnosis of CHD
Four articles were identified that examined SDH associations with
the fetal diagnosis of CHD (Table 2).
8–11
Three studies documen-
ted that poverty or low SES is associated with a significantly lower
likelihood of a prenatal CHD diagnosis; one study also found that
low maternal educational attainment and public insurance were
SDH risk factors for no prenatal CHD diagnosis. An analysis of 444
patients presenting to Boston Children’s Hospital with critical CHD
(defined as surgical or catheter intervention required in infants
≤30 days old) revealed that only 35% of those in the lowest SES
composite-score quartile received a prenatal CHD diagnosis, vs.
62% of those in the highest SES quartile. A retrospective study of
>2.5 million infants born in California revealed that the lowest
income tertile, public insurance, and low maternal educational
attainment were associated with a significantly higher likelihood
of CHD. In a study of 535 patients presenting to Children’s Hospital
of Wisconsin with CHD, residing in a higher poverty zip code was
associated with a significantly lower odds of a prenatal CHD
diagnosis. One study of 100 Cincinnati infants with CHD found no
association of family income, parental educational attainment, or
insurance coverage with prenatal CHD diagnosis, although the
sample size (100) was limited and multivariable analyses were not
performed.
CHD incidence and prevalence
Fifteen articles examined the association of SDH with CHD
incidence and prevalence (Table 2).
12–26
Poverty generally was
found to be significantly associated with CHD incidence and
prevalence, but such associations were either equivocal or lacking
for other SDH examined, including food insecurity, immigration,
and parental educational attainment.
Eleven articles analyzed an association between low SES and an
increased CHD incidence or prevalence, and most found that SES
was significantly associated with CHD incidence or prevalence. SES
definitions, however, varied among the studies, with low SES
defined as individual poverty, low family income, neighborhood
poverty, maternal educational attainment, parental employment,
operator/laborer occupation, crowding, rental occupancy, or some
combination thereof.
12–15,17–21,25,26
A study of 1.9 million children
born in Ontario, Canada, revealed that birth in low SES areas was
associated with significantly higher CHD rates (rate ratio =1.20;
95% confidence interval [CI] =1.15–1.24).
12
A population-based
study of 2.4 million live-born infants in California documented that
those residing in neighborhoods with the lowest SES composite
score had a significantly higher CHD incidence vs. those from the
highest SES neighborhoods (adjusted odds ratio (OR) =1.31; 95%
CI, 1.21–1.41).
25
Low SES also was found to be associated with a
significantly higher CHD incidence risk in studies using cardiology
clinic registries
13
and national databases.
17
Social determinants of health and outcomes for children and adults with.. .
B Davey et al.
2
Pediatric Research _#####################_
Table 1. Strategies for database searches (up to May 18, 2020).
Database Search strategy Hits
PubMed/Medline
(including Pre-MEDLINE
and non-MEDLINE)
(“Heart Defects, Congenital”[Mesh] OR “congenital heart”[tw] OR “congenital cardiac”[tw] OR
“cardiac anomaly”[tw] OR “cardiac anomalies”[tw] OR “conotruncal heart defects”[tw] OR
dextrocardia[tw] OR “double outlet right ventricle”[tw] OR “ectopia cordis”[tw] OR “fontan
procedure”[tw] OR “glenn procedure”[tw] OR “tetralogy of fallot”[tw] OR “tricuspid
atresia”[tw] OR “univentricular heart”[tw]) AND (“Social Determinants of Health”[Mesh] OR
“Socioeconomic Factors”[Mesh] OR socioeconomic[tw] OR socio-economic[tw] OR
sociodemographic[tw] OR socio-demographic[tw] OR social[tw] OR economic[tw] OR
poverty[tw] OR income[tw] OR financial[tw] OR employment[tw] OR unemployment[tw] OR
“marital status”[tw] OR “education level”[tw] OR “educational level”[tw] OR “education
status”[tw] OR “educational status”[tw] OR “Nutritional Status”[Mesh] OR “food
insecurity”[tw] OR “Healthcare Disparities”[Mesh] OR disparit*[tw] OR insurance[tw] OR
uninsured[tw] OR “Social Environment”[Mesh] OR “population density”[tw] OR “Residence
Characteristics”[Mesh] OR “deprived areas”[tw] OR housing[tw] OR residential[tw] OR
residence[tw] OR urban[tw] OR rural[tw] OR “Communication Barriers”[Mesh] OR “Emigrants
and Immigrants”[Mesh] OR immigra*[tw] OR emigra*[tw] OR migrant[tw] OR
“Prejudice”[Mesh] OR prejudic*[tw] OR “Domestic Violence”[Mesh] OR abuse[tw] OR
“Substance-Related Disorders”[Mesh] OR addict[tw] OR addict*[tw] OR alcohol[tw] OR
“Smoking”[Mesh] OR smok*[tw] OR “Tobacco Smoke Pollution”[Mesh]) AND (“United
States”[Mesh] OR “united states”[tw] OR USA[tw] OR Alabama[tw] OR Alaska[tw] OR Arizona
[tw] OR Arkansas[tw] OR California[tw] OR Colorado[tw] OR Connecticut[tw] OR Delaware
[tw] OR “District of Columbia”[tw] OR Florida[tw] OR Georgia[tw] OR Hawaii[tw] OR Idaho[tw]
OR Illinois[tw] OR Indiana[tw] OR Iowa[tw] OR Kansas[tw] OR Kentucky[tw] OR Louisiana[tw]
OR Maine[tw] OR Maryland[tw] OR Massachusetts[tw] OR Michigan[tw] OR Minnesota[tw] OR
Mississippi[tw] OR Missouri[tw] OR Montana[tw] OR Nebraska[tw] OR Nevada[tw] OR “New
Hampshire”[tw] OR “New Jersey”[tw] OR “New Mexico”[tw] OR “New York”[tw] OR “North
Carolina”[tw] OR “North Dakota”[tw] OR Ohio[tw] OR Oklahoma[tw] OR Oregon[tw] OR
Pennsylvania[tw] OR “Rhode Island”[tw] OR “South Carolina”[tw] OR “South Dakota”[tw] OR
Tennessee[tw] OR Texas[tw] OR Utah[tw] OR Vermont[tw] OR Virginia[tw] OR Washington[tw]
OR “West Virginia”[tw] OR Wisconsin[tw] OR Wyoming[tw] OR “Canada”[Mesh] OR canada
[tw] OR Alberta[tw] OR “British Columbia”[tw] OR Manitoba[tw] OR “New Brunswick”[tw] OR
Newfoundland[tw] OR Labrador[tw] OR “Northwest Territories”[tw] OR “Nova Scotia”[tw] OR
Nunavut[tw] OR Ontario[tw] OR “Prince Edward Island”[tw] OR Quebec[tw] OR Saskatchewan
[tw] OR “Yukon Territory”[tw] OR “united states”[ad] OR USA[ad] OR Alabama[ad] OR Alaska
[ad] OR Arizona[ad] OR Arkansas[ad] OR California[ad] OR Colorado[ad] OR Connecticut[ad]
OR Delaware[ad] OR “District of Columbia”[ad] OR Florida[ad] OR Georgia[ad] OR Hawaii[ad]
OR Idaho[ad] OR Illinois[ad] OR Indiana[ad] OR Iowa[ad] OR Kansas[ad] OR Kentucky[ad] OR
Louisiana[ad] OR Maine[ad] OR Maryland[ad] OR Massachusetts[ad] OR Michigan[ad] OR
Minnesota[ad] OR Mississippi[ad] OR Missouri[ad] OR Montana[ad] OR Nebraska[ad] OR
Nevada[ad] OR “New Hampshire”[ad] OR “New Jersey”[ad] OR “New Mexico”[ad] OR “New
York”[ad] OR “North Carolina”[ad] OR “North Dakota”[ad] OR Ohio[ad] OR Oklahoma[ad] OR
Oregon[ad] OR Pennsylvania[ad] OR “Rhode Island”[ad] OR “South Carolina”[ad] OR “South
Dakota”[ad] OR Tennessee[ad] OR Texas[ad] OR Utah[ad] OR Vermont[ad] OR Virginia[ad] OR
Washington[ad] OR “West Virginia”[ad] OR Wisconsin[ad] OR Wyoming[ad] OR canada[ad]
OR Alberta[ad] OR “British Columbia”[ad] OR Manitoba[ad] OR “New Brunswick”[ad] OR
Newfoundland[ad] OR Labrador[ad] OR “Northwest Territories”[ad] OR “Nova Scotia”[ad] OR
Nunavut[ad] OR Ontario[ad] OR “Prince Edward Island”[ad] OR Quebec[ad] OR Saskatchewan
[ad] OR “Yukon Territor y”[ad] OR AL[ad] OR AK[ad] OR AZ[ad] OR AR[ad] OR CA[ad] OR CO
[ad] OR CT[ad] OR DE[ad] OR DC[ad] OR FL[ad] OR GA[ad] OR HI[ad] OR ID[ad] OR IL[ad] OR
IN[ad] OR IA[ad] OR KS[ad] OR KY[ad] OR LA[ad] OR ME[ad] OR MD[ad] OR MA[ad] OR MI[ad]
OR MN[ad] OR MS[ad] OR MO[ad] OR MT[ad] OR NE[ad] OR NV[ad] OR NH[ad] OR NJ[ad] OR
NM[ad] OR NY[ad] OR NC[ad] OR ND[ad] OR OH[ad] OR OK[ad] OR Ore[ad] OR PA[ad] OR RI
[ad] OR SC[ad] OR SD[ad] OR TN[ad] OR TX[ad] OR UT[ad] OR VT[ad] OR VA[ad] OR WA[ad]
OR WV[ad] OR WI[ad] OR WY[ad] OR AB[ad] OR BC[ad] OR MB[ad] OR NB[ad] OR NL[ad] OR
NT[ad] OR NS[ad] OR NU[ad] OR ON[ad] OR PE[ad] OR QC[ad] OR SK[ad] OR YT[ad]) NOT
“Marfan Syndrome”[Mesh] NOT “Myocarditis”[Mesh] NOT (“Animals”[Mesh] NOT
(“Animals”[Mesh] AND “Humans”[Mesh])) NOT (“Editorial”[pt] OR “Letter”[pt] OR “Case
Reports”[pt] OR “Systematic Review”[pt] OR “Meta-Analysis”[pt] OR “Comment”[pt])
1845
Scopus (Elsevier) TITLE-ABS-KEY((“congenital heart”OR “congenital cardiac”OR “cardiac anomaly”OR
“conotruncal heart defects”OR dextrocardia OR “double outlet right ventricle”OR “ectopia
cordis”OR “fontan procedure”OR “glenn procedure”OR “tetralogy of fallot ”OR “tricuspid
atresia”OR “univentricular heart”) AND (socioeconomic OR socio-economic OR
sociodemographic OR socio-demographic OR social OR economic OR poverty OR income
OR financial OR employment OR unemployment OR “marital status”OR “education level”OR
“educational level”OR “education status”OR “educational status”OR “food insecurity”OR
disparit* OR insurance OR uninsured OR “population density”OR “deprived areas”OR
impoverished OR housing OR residential OR residence OR urban OR rural OR immigra* OR
emigra* OR migrant OR prejudic* OR abuse OR violence OR addict OR addict* OR alcohol OR
smok*) AND (“united states”OR usa OR alabama OR alaska OR arizona OR arkansas OR
california OR colorado OR connecticut OR delaware OR “District of Columbia”OR florida OR
georgia OR hawaii OR idaho OR illinois OR indiana OR iowa OR kansas OR kentucky OR
louisiana OR maine OR maryland OR massachusetts OR michigan OR minnesota OR
mississippi OR missouri OR montana OR nebraska OR nevada OR “New Hampshire”OR “New
Jersey”OR “New Mexico”OR “New York”OR “North Carolina”OR “North Dakota”OR ohio OR
oklahoma OR oregon OR pennsylvania OR “Rhode Island”OR “South Carolina”OR “South
Dakota”OR tennessee OR texas OR utah OR vermont OR virginia OR washington OR “West
Virginia”OR wisconsin OR wyoming OR canada OR alberta OR “British Columbia”OR
687
Social determinants of health and outcomes for children and adults with. . .
B Davey et al.
3
Pediatric Research _#####################_
Two articles used the Nationwide Inpatient Sample (NIS) to
examine secular trends in CHD prevalence, but reached different
conclusions. One study demonstrated that those in the upper
income quartile experienced a significantly greater temporal
decrease in the prevalence of severe CHD vs. those in the lowest
income quartile,
19
whereas another found that mild CHD
prevalence significantly increased only in the high SES group.
20
Another population-based study using the NIS reported that the
overall CHD incidence was actually significantly lower in the
lowest SES group, although the authors speculated that this may
have been due to lower access to hospitals with better diagnostic
tools.
21
A study on food insecurity as a risk factor for conotruncal heart
defects reported that food insecurity was associated with higher
adjusted odds of D-transposition of the great arteries, but only
among normal-weight and underweight mothers (and not those
who were overweight or obese); no association of food insecurity,
however, was found with tetralogy of Fallot.
16
An analysis of the
National Birth Defects Prevention Study revealed that having
immigrant parents was associated with significant lower odds of
certain CHDs, with the greatest number of significantly protective
adjusted ORs noted for foreign-born parents residing ≤5 years vs.
>5 years in the US.
22
Another study, however, found no
association of maternal birthplace with left ventricular outflow-
tract malformations.
24
Two studies found no association of
maternal educational attainment with CHD prevalence.
23,24
Infant mortality
Nine articles analyzed associations between SDH and infant
mortality in CHD patients (Table 2). Poverty, low parental
educational attainment, uninsurance, transportation barriers, and
immigration status were significantly associated with infant
mortality.
1–3,27–32
A study of 229 children with hypoplastic left heart syndrome
(HLHS) identified via the Metropolitan Atlanta Congenital Defects
Program documented survival rates that were almost three times
Table 1. continued
Database Search strategy Hits
manitoba OR “New Brunswick”OR newfoundland OR labrador OR “Northwest Territories”OR
“Nova Scotia”OR nunavut OR ontario OR “Prince Edward Island”OR quebec OR
saskatchewan OR “Yukon Territory”OR (affilcountry, “United States”) OR (affilcountry,
“Canada”)) AND NOT marfan AND NOT myocarditis) AND (LIMIT-TO (DOCTYPE, “ar”))
Cochrane Central
Register of Controlled Trials
(CENTRAL) (Wiley)
All Text: (“congenital heart”OR “congenital cardiac”OR “cardiac anomaly”OR “conotruncal
heart defects”OR dextrocardia OR “double outlet right ventricle”OR “ectopia cordis”OR
“fontan procedure”OR “glenn procedure”OR “tetralogy of fallot”OR “tricuspid atresia”OR
“univentricular heart”) AND (socioeconomic OR socio-economic OR sociodemographic OR
socio-demographic OR social OR economic OR poverty OR income OR financial OR
employment OR unemployment OR “marital status”OR “education level”OR “
educational
level”OR “education status”OR “educational status”OR “food insecurity”OR disparit* OR
insurance OR uninsured OR “population density”OR “deprived areas”OR impoverished OR
housing OR residential OR residence OR urban OR rural OR immigra* OR emigra* OR migrant
OR prejudic* OR abuse OR violence OR addict OR addict* OR alcohol OR smok*) NOTmarfan
NOT myocarditis
175
CINAHL (Ebsco) (MH “Heart Defects, Congenital+”OR TX “congenital heart”OR TX “congenital cardiac”OR
TX “cardiac anomaly”OR TX “conotruncal heart defects”OR TX dextrocardia OR TX “double
outlet right ventricle”OR TX “ectopia cordis”OR TX “fontan procedure”OR TX “glenn
procedure”OR TX “tetralogy of fallot”OR TX “tricuspid atresia”OR TX “univentricular heart”)
AND (MH “Social Determinants of Health”OR MH “Socioeconomic Factors+”OR TX
socioeconomic OR TX socio-economic OR TX sociodemographic OR TX socio-demographic
OR TX social OR TX economic OR TX poverty OR TX income OR TX financial OR TX
employment OR TX unemployment OR MH “Marital Status+”OR TX “marital status”OR TX
“education level”OR TX “educational level”OR TX “education status”OR TX “educational
status”OR MH “Nutritional Status”OR MH “Food Security”OR TX “food insecurity”OR MH
“Healthcare Disparities”OR TX disparit* OR TX insurance OR TX uninsured OR MH “Social
Environment+”OR MH “Population Density”OR TX “population density”OR TX “deprived
area”OR TX “deprived areas”OR TX impoverished OR MH “Residence Characteristics+”OR
TX housing OR TX residential OR TX residence OR TX urban OR TX rural OR MH
“Communication Barriers”OR TX “non-English-speaking”OR MH “Immigrants+”OR TX
immigra* OR TX emigra* OR TX migrant OR MH “Prejudice”OR TX prejudic* OR MH
“Domestic Violence+”OR TX abuse OR TX violence OR MH “Substance Dependence+”OR
TX addict OR TX addict* OR TX alcohol OR TX smok*) AND (MH “United States+”OR MH
“Canada+”) NOT TX marfan NOT TX myocarditis
Limit to: Human
Limit to Geographic Subset: Canada and USA
408
PsycInfo (Ebsco) (“congenital heart”OR “congenital cardiac”OR “cardiac anomaly”OR “conotruncal heart
defects”OR dextrocardia OR “double outlet right ventricle”OR “ectopia cordis”OR “fontan
procedure”OR “glenn procedure”OR “tetralogy of fallot”OR “tricuspid atresia”OR
“univentricular heart”) AND (socioeconomic OR socio-economic OR sociodemographic OR
socio-demographic OR social OR economic OR poverty OR income OR financial OR
employment OR unemployment OR “marital status”OR “education level”OR “educational
level”OR “education status”OR “educational status”OR “food insecurity”OR disparit* OR
insurance OR uninsured OR “population density”OR “deprived areas”OR impoverished OR
housing OR residential OR residence OR urban OR rural OR immigra* OR emigra* OR migrant
OR prejudic* OR abuse OR violence OR addict OR addict* OR alcohol OR smok*) NOTmarfan
NOT myocarditis
294
SocIndex (Ebsco) (TX “congenital heart”OR TX “congenital cardiac”OR TX “cardiac anomaly”) NOT TX marfan
NOT TX myocarditis
583
Social Interventions
Research & Evaluation Network
(SIREN) Evidence & Resource Library
(University of California, San Francisco)
Text Searches: “congenital hear t”,“congenital cardiac”,“cardiac anomaly”,“conotruncal heart
defects”, dextrocardia, “double outlet right ventricle”,“ectopia cordis”,“fontan procedure”,
“glenn procedure”,“tetralogy of fallot”,“tricuspid atresia”,“univentricular heart”
0
Social determinants of health and outcomes for children and adults with.. .
B Davey et al.
4
Pediatric Research _#####################_
worse for those residing in high-poverty (9%) vs. low-poverty
(25%; P< 0.001) neighborhoods.
32
An analysis of data from birth-
defect surveillance programs from four states (Arizona, New York,
New Jersey, and Texas) on almost 10,000 infants with CHD
revealed that poverty was associated with about double the
adjusted odds of infant mortality.
3
Low maternal educational attainment was associated with a
significantly higher risk of CHD infant mortality in three
studies.
1,3,30
For example, one study of coarctation of the aorta
revealed a mortality rates of 27% for infants of mothers who had
not completed high school vs. 5% for those who at least
completed high school (P=0.004).
30
Research on 4390 infants
with CHD also documented that lower paternal educational was
associated with a 62% increased risk of infant mortality.
31
An analysis of the Texas Birth Defects Registry revealed that
uninsured infants with critical and noncritical CHDs had approxi-
mately triple and double the risk of neonatal mortality,
respectively, compared with infants with private insurance.
29
Another Texas study found that residing in a county bordering
Mexico was associated with higher adjusted odds of CHD infant
mortality.
28
Post-surgical outcomes
A total of 25 articles evaluated the association of SDH with post-
surgical outcomes in CHD patients (Table 2). Poverty and low SES
were consistently associated with adverse post-operative out-
comes, including worse HLHS survival,
33
increased in-hospital
mortality and resource utilization after orthotopic heart transplant
for single-ventricle vs. cardiomyopathy patient cohorts,
34
higher
inter-stage mortality in the Single Ventricle Reconstruction Trial,
35
higher mortality following congenital heart surgery,
36
worse 1-
year transplant-free survival after the Norwood procedure (stage I
palliation for single-ventricle CHD),
37
unplanned readmission in
the first 90 days after congenital heart surgery,
38
longer length of
Records identified through database
searching
(n = 3992)
PubMed/MEDLINE (n = 1845)
Scopus (n = 687)
Cochrane Central Register of Controlled Trials (n = 175)
CINAHL (n = 408)
PsycInfo (n = 294)
SocIndex (n = 583)
SIREN (n = 0)
Additional records identified
through other sources
(n = 0)
Records excluded (n = 3061)*
Records excluded (n = 31)*
1. Structural CHD not focus of
investigation (n = 924)
2. SDH not assessed in analysis (n = 1155)
3. Both 1 + 2 (n = 328)
5. Publications outside United States and
Canada (n = 329)
6. Duplicate (n = 15)
1. Structural CHD not focus of
investigation (n = 2)
2. SDH not assessed in analysis (n = 14)
3. Structural CHD not focus of
investigation and SDH not assessed in
analysis (n = 1)
5. Publications outside United States and
Canada (n = 7)
4. Letter to editor, commentaries,
editorials, viewpoint, perspectives,
opinion pieces, case reports, book
chapter, author or keyword index or
review article (n = 7)
4. Letter to editor, commentaries,
editorials, viewpoint, perspectives,
opinion pieces, case reports, book
chapter, author or keyword index or
review article (n = 310)
Records after duplicates removed
(n = 3180)
IdentificationScreeningEligibilityIncluded
Records screened by title/abstract
(n = 3180)
Full-text articles assessed
for eligibility
(n = 119)
Studies included in
qualitative synthesis
(n = 88)
*Studies may have been excluded for multiple reasons
Fig. 1 Flow diagram of study selection (adapted from PRISMA), including study identification, screening, eligibility, and final inclusion
procedures and numbers.
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5
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Table 2. Summary of the included studies on the association of social determinants of health with congenital heart disease (CHD).
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
Fetal diagnosis of congenital heart disease—4
Poverty/socioeconomic status (SES)
—Patients with public insurance, lower SES,
and living below the poverty level less likely to
have a prenatal diagnosis of critical CHD
Retrospective analysis of infants presenting to
the Boston Children’s with critical CHD from
2003 to 2006
N=444 Adjusted for 4 covariates
9
—Peiris
Poverty/transportation barriers
—Maternal educational attainment, income,
and insurance type not significantly associated
with prenatal diagnosis
Prospective study of 100 consecutive infants
diagnosed with major CHD who received
prenatal care in Cincinnati eight-county area
from October 2007 to May 2009
N=100 Adjusted for 7 covariates
11
—Sekar
Poverty/transportation barriers
—Those living in impoverished or rural
communities were at highest risk of not
having diagnosis made prenatally
Patients presenting to the Children’s Hospital
of Wisconsin with critical CHD from 2007
to 2013
N=535 Adjusted for 9 covariates
8
—Hill
Poverty/parental educational attainment
—Infants with critical CHD were more likely to
be born to mothers enrolled in WIC and have
public insurance and less likely to be born to
mothers with college degrees or mother at
top SES tertile
Linked dataset from the Office of Statewide
Health Planning and Development to access
ICD-9 diagnosis codes for all infants born in
California from 2008 to 2012 to compare
infants with critical CHD to general population
N=2,514,837 Adjusted for 12 covariates
10
—Purkey
CHD prevalence and incidence—15
Poverty/parental educational attainment
—For whites in the lowest SES stratum, risk of
aortic stenosis was five times that of blacks
—Controlling for socioeconomic factors
attenuated the white excess for Ebstein’s
anomaly and disclosed white excess for L-TGA
Population-based case-control study in
Maryland, the District of Columbia, and
northern Virginia between 1981 and 1987
N=4808 (n=2087 cases, n=2721
controls)
Adjusted for 6 covariates, including maternal
educational attainment, occupation of the
head of the household, and family income
per family member
18
—Correa-
Villaseñor
Low income
—Medicaid coverage associated with CHD
higher rate
The University of Arizona Pediatric Cardiology
Registry between 1990 and 1994.
Questionnaire to all mothers to confirm data
collected from patient records and maternal
demographics
N=831 Chi-square test, no covariates
13
—Baron
Poverty/parental educational attainment
—Low SES associated with increased risk of D-
transposition of the great arteries (dTGA),
reduced risk of tetralogy of Fallot (TOF)
(prevalence)
Interview data from case mothers and a
population-based case–control study in Los
Angeles, San Francisco, and Santa Clara from
1987 to 1989
N=1430 (n=734 control, n=207
conotruncal cases, n=348 isolated
cleft lip with/without cleft palate, n
=141 isolated cleft lip without cleft
palate)
Adjusted for 4 covariates
Individual SES defined by maternal education
and parental employment; neighborhood
SES defined by education, poverty,
unemployment, operator/laborer occupation,
crowding, and rental occupancy
15
—Carmichael
Educational attainment/immigration status
—Maternal educational attainment and
birthplace not significantly associated with
prevalence rate ratios for left ventricular
outflow tract
The Texas Birth Defect Registry database from
1999 to 2001
N=1,077,574 Adjusted for 5 covariates
24
—McBride
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
Food insecurity
—Food insecurity associated with higher
adjusted odds of D-transposition of the great
arteries but only among normal-weight and
underweight mothers (and not those who
were overweight or obese); no association of
food insecurity, however, with tetralogy of
Fallot
Population base case–control data in Los
Angeles, San Francisco, and Santa Clara
counties from 1999 to 2004
N=1884 (n=1189 cases; n=695
controls)
Adjusted for 7 covariates
16
—Carmichael
Low SES/parental educational attainment
—Operator/laborer mothers at two times
greater risk to have offspring with TOF and
dTGA (AOR ≥2.2), and either parent
unemployed was more likely to have dTGA
cases (AOR ≥1.4)
Data from National Birth Defect Prevention
Study in 1997–2000
N=4392 (n=2551 controls and
n=1841 cases)
Adjusted for 8 covariates
SES—measured by parents’educational
attainment, occupation, and
household income
26
—Yang
Poverty/parental educational attainment
—Low maternal educational attainment and
low income not associated with increased risk
of dTGA
—Low income not associated with increased
risk of TOF
Population-based case–control study in
California from July 1999 to June 2004
N=1502 (n=617 non-malformed
controls, n=608 with orofacial
clefts, n=277 with conotruncal
heart defects)
Adjusted for 5 covariates
SES was defined by maternal educational
attainment, maternal and paternal
employment, and annual household income
14
—Carmichael
Parental educational attainment/
immigration status
—Maternal residence along Texas–Mexico
border associated with increase in prevalence
of isolated TA
—No significant association between low
maternal educational attainment and
increased prevalence of dTGA or TOF
Texas Birth Defects Registry from 1999 to 2004 N=2,208,758 “Variables that were significantly related to
the outcome of interest in the crude analyses
and for which <10% of cases had missing
values were included in the multivariate
analyses”
23
—Long
Low SES/parental educational attainment
—Children born in low SES areas had
significantly higher CHD prevalence
Healthcare data through Ontario Ministry of
Health and Long-Term Care (MOHLTC)—all
children born alive in hospital in Ontario,
Canada, 1994–2007
N=1,871,760 Adjusted for 3 covariates
SES defined by neighborhood income and
educational attainment
12
—Agha
Poverty
—Incidence of CHD similar for all SES classes
except lowest SES class, which had
significantly lower CHD incidence
The Nationwide Inpatient Sample from Jan
2008 to Dec 2008
N=1,204,887 Descriptive statistics
21
—Egbe
Poverty
—Prevalence of TOF, TA, HLHS, and PA from
1999 to 2008 did not change significantly in
the lowest income quartile
—In contrast, significant temporal decrease in
prevalence of TOF, TA, HLHS, and PA in the
highest income quartile, decrease in the
prevalence of PA in the third income quartile,
and decrease in the prevalence of TOF, TA, and
HLHS in second income quartile (authors
speculate that this may be due to increased
prenatal diagnosis of CHD and termination of
pregnancy)
Population-based study—the Nationwide
Inpatient Sample from 1999 to 2008
N=9,696,908 Descriptive statistics
19
—Egbe
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
Low income
—Prevalence of isolated PDA increased across
all income quartiles from 1998 to 2008
—No change in prevalence of mild CHD in
lowest income quartiles; increase in
prevalence of mild CHD in highest income
quartile (authors speculate that this may be
due to access to hospitals with better
diagnostic tools, although could be influenced
by missing data)
The Nationwide Inpatient Sample in 1998 and
in 2008
N=1,990,893 Descriptive statistics
20
—Egbe
Immigration status
—Infants with both parents foreign-born had
lower unadjusted rates of six CHDs
—Infants with foreign-born mothers had
lower crude rate of aortic stenosis
—After adjustment, immigrant protective
associations remained for aortic stenosis, right
ventricular outflow tract obstruction, and
pulmonary valve stenosis
—more statistically significant protective
results observed in infants with parents who
had spent ≤5 years in US
National Birth Defects Prevention Study,
1997–2011
N=44,029 (n=32,200 cases,
n=11,829 controls)
Adjusted for 11 covariates
22
—Hoyt
Low income
—Low income associated with CHD
prevalence; compared with infants of diabetic
mothers born in family with highest 25th
quartile family income, infants in lowest 25th
quartile family income had higher
odds of CHD
Nationally representative Kids’Inpatient
Database (KID) for years 2003, 2006, 2009,
and 2012
N=183,453 Adjusted for 1 covariate (race/ethnicity) or
unadjusted
17
—Chou
SES/parental educational attainment
—Lower SES at neighborhood level associated
with incidence of live-born CHD and was most
significant for those with the highest social
deprivation
Population-based cohort study in California
(2007–2012)
Socioeconomic variables for each subject’s
census tract collected from US Census website
N=2,419,651 Adjusted for 2 covariates
Social deprivation index [SDI] determined at
the neighborhood level
SES defined by income, parental educational
attainment, occupation at
neighborhood level
25
—Peyvandi
Infant mortality—9
Poverty/parental educational attainment
—Low paternal educational attainment
associated with increased mortality of infants
with CHD, whether diagnosis was made before
or after death
—Lower income, maternal educational
attainment, and SES scores not associated
with death before CHD diagnosis
Infants with CHD identified in a population-
based study between 1981 and 1989 in the
Baltimore Washington metropolitan area
N=4390 Adjusted for 6 covariates
30
—Kuehl
Uninsured/parental educational attainment/
poverty
—Death rates 33 vs. 4% for infants of
uninsured vs. insured mothers for coarctation
of aorta
Baltimore Washington Infant Study of infantile
coarctation of aorta
N=105 Adjusted for 5 covariates
31
—Kuehl
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
—27.3% mortality for infants of mothers who
had not completed high school vs. 4.9% for
those who at least completed high school
(P=0.004)
—Income and paternal educational
attainment not associated with survival in
multivariate analysis
Poverty/immigration status/transportation
barriers
—Lower first-year survival and lower referral
rate in border counties (bordering Mexico)
—Maternal educational attainment, distance
to a major center, and foreign-born parents
not associated with mortality
Infants with severe CHD, born 1996–2003,
identified from Texas Birth Defects Registry
N=1213 Adjusted for 12 covariates
28
—Fixler
Low income/uninsured
—Uninsured infants with critical and
noncritical CHDs had approximately 3 and 2
times the increased neonatal mortality risk,
respectively, vs. infants with private insurance
—Publicly insured infants had 30% reduced
mortality risk vs. privately insured infants
during neonatal period, but 30% increased risk
in the post-neonatal period
Population-based, retrospective study in a
cohort of Florida resident infants born with
CHDs between 1998 and 2007
N=43,411 Adjusted for 8 covariates
29
—Kucik
Poverty/parental educational attainment
—Census-tract-level poverty and low parental
educational attainment associated with higher
adjusted odds of infant mortality
Population-based data from 4 state-based
birth defect surveillance programs (Arizona,
New York, New Jersey, and Texas)
in retrospective cohort study of infants from
1999 to 2007 with CHD
N=10,578 Adjusted for 11 covariates
3
—Kucik
Poverty
—High neighborhood poverty associated with
higher death rate and lower survival
probability of children with HLHS
Infants with nonsyndromic HLHS born
between 1979 and 2005 identified through
Metropolitan Atlanta Congenital Defects
Program
N=212 Adjusted for 10 covariates
32
—Siffel
Low income
—Deliveries covered by Medicaid (including
for infants with CHD) associated with infant
mortality attributable to birth defects
2011–2013 Linked Birth and Infant Death Data
from National Vital Statistics System for infants
<1 year old
N=9,542,603 Adjusted for 12 covariates
27
—Almli
Poverty/parental educational attainment
—Lower maternal educational attainment not
associated with increased hazard of infant
mortality
—Medicaid status not significantly associated
with increased or decreased hazard of infant
mortality, after adjustment for confounding
variables
Infants with CHDs born between 2004 and
2013 ascertained by NC Birth Defects
Monitoring Program
N=15,533 Adjusted for 22 covariates
2
—Pace
Poverty and educational attainment
—Lower maternal educational attainment and
public insurance had significantly increased
odds of poor outcome (mortality or re-
admission)
Population-based cohort study using
California Office of Statewide Health to assess
outcomes for live-born infants with HLHS
and dTGA
N=1796 Adjusted for 14 covariates
1
—Peyvandi
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
Post-surgical outcomes—23
Low income/transportation barriers
—Patients with Medicaid older at the time of
operation vs. patients with private insurance
and managed care
—No association of distance between
patient’s home and surgical center found for
ASD, VSD, TOF, or AVC
Office of Statewide Health Planning and
Development database for 1995 and 1996 in
California
N=666 No covariates; Kruskal–Wallis one-way
ANOVA, weighted linear regression
87
—Chang
Low income
—Publicly insured children significantly more
likely to use higher-mortality hospitals
for cardiac surgery vs. those with indemnity
coverage
Retrospective cohort study using annual
California state-mandated hospital discharge
dataset between 1992 and 1994
N=5071 Adjusted for 6 covariates
88
—Erickson
Low income
—Income not associated with treatment
choices
National Inpatient Sample dataset, 1998–1997 N=1986 Adjusted for 8 covariates
87
—Chang
Low income
—Medicaid coverage associated with higher
risk of dying after CHD surgery
Population-based retrospective cohort study
using hospital discharge abstract data from
five states (CA, MA, IL, PA, and WA) in 1992
and 1996
N=11,636 Adjusted for risk category (RACHS-1) and
additional clinical variables
38
—Lushaj
Low income/uninsured/transportation barrier
—Zip code median family income <$30,000
and distance between home and hospital
>100 miles associated with reduced likelihood
of readmission after neonatal cardiac surgery
—Uninsurance not significantly associated
with likelihood of readmission
Single center, case–control study at Boston
Children’s Hospital between 1992 and 2002
Case: N=498; control: N=254 Adjusted for 2 covariates
44
—Mackie
Low income
—Lower family income associated with lower
physical and psychosocial functioning
Pediatric Heart Network cross-sectional study
(pediatric cardiac centers in US and Canada)
N=537 Adjusted for 5 covariates
89
—McCrindle
Poverty
—Adjusting for median income by zip code
universally increased magnitude of ORs (of
death) and overall level of significance for
blacks and Latinos
Kids’Inpatient Database 2000 N=8483 Adjusted for 6 covariate
90
—Benavidez
Poverty
—Income not associated with post-
discharge death
Statewide hospital discharge data from
California 1989–1999
N=25,402 Adjusted for 1 covariate—“risk adjustment”
91,92
—Chang
Poverty/parental educational attainment/
transportation barriers
—Low SES associated with mental outcomes
—SES, maternal educational attainment, and
“location of family home”not associated with
growth and health outcomes
Interprovincial inception cohort study in
Western Canada from 1996 through 2004
N=41 No covariates. Descriptive analysis—chi-
square and Fisher’s exact test performed only
43
—Alton
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
Low income
—After adjustment for time since Fontan
procedure, multivariate linear regression
demonstrated that lower annual family
income associated with lower functional score,
indicating worse functional state
Pediatric Heart Network Fontan Cross-
Sectional Study completed in 2004 by 7
Network centers
N=476 Adjusted for 1 covariate (time since the
Fontan procedure)
93
—Williams
Poverty
—After adjusting for age/weight, infants with
HLHS in high-poverty areas 1.8 times (95% CI:
1.1–2.8; P=0.015) more likely to die than
those in high-poverty areas
Michigan Birth Defect Registry from 1992
to 2005
Control N=4060; case N =406 Adjusted for 2 covariates
33
—Hirsch
Low income
—Medicaid coverage associated with
mortality, non-elective admission for
congenital heart surgery, and referral to high-
mortality hospitals
Kids’Inpatient Database from 1997 to 2006 N=44,910 Adjusted for 5 covariates
94
—Chan
Parental educational attainment
—Lower maternal educational attainment
associated with lower MentaI Developmental
Index score (P=0.04)
SVR trial—15 centers in North America
between May 2005 and July 2008
N=373 Adjusted for 5 covariates
42
—Newburger
Low income
—Lower SES associated with intermediate-
term mortality
SVR trial between May 2005 and July 2008 at
15 centers in US and Canada
N=549 Adjusted for 7 covariates
95
—Tweddell
Poverty
—Census-block poverty associated with inter-
stage mortality (P=0.003)
—But subjects in communities with 5.4–13%
poverty had greater risk of inter-stage
mortality vs. subjects in poorest communities
(OR, 2.5)
Single Ventricle Reconstruction trial N=426 Adjusted for 4 covariates
35
—Ghanayem
Low income
—Low SES associated with lower physical
summary scores and school functioning scores
and decreased quality of life
Prospective cohort study at Stollery Children’s
hospital from July 2000 to June 2005
N=242 Adjusted for 8 covariates
Family SES determined by Blishen Index
41
—
Garcia Guerra
Low income
—Lower income quartiles associated with
surgical ligation
—Payer status and income quartile not
associated with survival
Kids’Inpatient Data from the 1997, 2000, 2003,
2006, and 2009 releases
N=63,208 Adjusted for covariates; regression adjusted
for comorbid risk factors using Elixhauser
method, which has been validated in
multiple previous studies
37
—Tashiro
Low income
—Public insurance associated with longer
length of stay and increased in-hospital
neonatal mortality
2012 Healthcare Cost and Utilization Project
Kids Inpatient Database
N=13,130 Multivariable logistic regression with sample
weights, stratification, and clustering
40
—Peterson
Low income/educational attainment
—After adjustment for patient demographics,
birth characteristics, and anatomy, patients in
lowest SES tertile had significantly higher risk
of death or transplant than patients in highest
SES tertile
Pediatric Heart Network Single Ventricle
Reconstruction (SVR) Trial Public Use dataset
between May 2005 and July 2008 at 15 centers
in US and Canada
N=525 Adjusted for 6 covariates
96
—Bucholz
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
—Low neighborhood SES associated with
worse 1-year transplant-free survival after
Norwood procedure
Poverty
—income not significantly associated with risk
for readmission in adjusted analysis
State Inpatient Databases for Washington,
New York, Florida, and California
N=8585 Adjusted for 7 covariates
97
—Benavidez
Low income
—Lower median household income
associated with increased resource utilization
and lower in-hospital mortality in single-
ventricle CHD patients who undergo OHT
PHIS database between January 2004 and
September 2015
N=1599 Adjusted for 12 covariates
34
—Bradford
Low income
—Low income associated with difficulties in
adaptive behavior, behavioral symptoms, QOL,
and functional status in children with
hypoplastic left heart syndrome at 6 years old
Single Ventricle Reconstruction Trial from 2005
to 2008
N=250 Adjusted for 7 covariates
98
—Goldberg
Low income/transportation barrier
—Lower SES risk factor for unplanned
readmissions in first 90 days after surgery
—Distance to hospital inversely associated
with readmissions (P< 0.01), with odds of
patient getting readmitted decreasing by 33%
for each 100 miles of distance lived farther
from the hospital
Retrospective review of patients at University
of Wisconsin School of Medicine and Public
Health, August 2011–June 2015
N=265 No covariates.
Bivariable logistic regression, general
linear model
38
—Lushaj
Access to care, loss to follow-up, and hospital readmissions—10
Low income
—Medicaid coverage associated with high
resource utilization
Review of hospital discharge data from
Healthcare Cost and Utilization Project (HCUP)
Kids’Inpatient Database (KID) year 2000 (data
from 27 states) for patients <18 years old who
had congenital heart surgery
N=10,569 Adjusted for 30 covariates
39
—Connor
Low income
—Lower median family income associated
with increased risk of loss to follow-up
(adjusted odds of 1.2 per $10,000 decrement
in income)
Matched case-control design to examine risk
factors for loss to cardiology follow-up among
children and young adults with CHD using
Western Canadian Children’s Heart Network
database
N=296 Adjusted for 12 covariates
47
—Mackie
Low SES (public insurance)
—Significant trend over time in proportion of
publicly insured admissions (42–61%; P<
0.0001) to and bed days in pediatric
cardiology specialty-care centers from 1983
to 2011
Retrospective analysis of pediatric CHD
patients seen at pediatric cardiology specialty-
care centers from 1983 to 2011, using
California Office of Statewide Health Planning
and Development unmasked database
N=164,310 Adjusted for 6 covariates
99
—
Chamberlain
Low income
—Public insurance and lower household
income (<$52,950) associated with/missed
appointments in bivariate analysis
Single-center retrospective study of patients
with outpatient congenital or pediatric cardiac
MR appointments from January 1, 2014,
through December 31, 2015
N=795 No multivariable adjustment for covariates
46
—Lu
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
Poverty/transportation barriers
—Significant interaction between region
rurality and census-tract poverty in
multivariate analysis of one-way drive-time
predictors
Population-based, 11-county surveillance
system of CHDs in New York, to characterize
proximity to nearest pediatric cardiac surgical
care center among adolescents 11–19 years
old with CHDs
N=2522 Adjusted for 9 covariates
49
—
Sommerhalter
Poverty/educational attainment/
transportation barriers
—Median income below 25th percentile (P=
0.03) and less than college education (P=
0.03) associated with non-attendance at
neurodevelopmental follow-up clinic
—Residing ≥200 miles from surgical center
show non-significant trend for non-
attendance in multivariate analysis (adjusted
OR, 2.86; P=0.054)
—Public insurance associated with non-
attendance (P=0.01)
Single center retrospective review of survivors
of infant (<1 year old) cardiac surgery (4/
2011–3/2014) to examine prevalence of
neurodevelopmental evaluation
N=552 Adjusted for 20 covariates
48
—Loccoh
Low income (public insurance)
—Medicaid coverage associated with 1.2
adjusted odds of lapsing in (missing) yearly
cardiology follow-ups
Single center retrospective review of CHD
patients with moderate to severe complexity
in large, urban pediatric hospital in Midwest
between 2007 and 2011
N=1034 Adjusted for 6 covariates
45
—Jackson
Low income
—Cumulative social risk associated with
readmission days for CHD patients with low
risk of morbidity by procedure
Single center retrospective review of patients
who underwent infant cardiac surgery
with CPB
N=219 Adjusted for 6 covariates
100
—
Demianczyk
Low income/transportation barriers
—Among publicly insured infants with CHD
who require early surgery, many live far away
from surgical centers that can provide
definitive care, with some demographic and
geographic groups at a particular
disadvantage
2012 Medicaid Analytic eXtract data from
40 states reviewed for infants with CHD
requiring surgery in first year of life
N=4598 Adjusted for 13 covariates
101
—Woo
Immigration status
—Higher appointment no-show rate (16–20%)
in cardiology outreach clinic targeting
immigrant and resettled refugee community,
compared with national benchmark of <10%
Data obtained between 2014 and 2017 from a
monthly pediatric cardiology clinic at a
Federally Qualified Health Center
N=366 Observational study
No statistical analysis
50
—Agrawal
Neurodevelopmental outcomes and QOL—8
Poverty
—Family’s SES not associated with
parental stress
Abidin’s Parenting Stress Index administered
to parents of children 2–12 years old with CHD
N=80 Adjusted for 7 covariates
55
—Uzark
Parental educational attainment
—Lower maternal educational attainment
associated with worse outcomes for
performance IQ, socialization, adaptive
Prospective study of infants with CHDs who
underwent surgical repair in infancy
N=94 Adjusted for 8 covariates
52
—Majnemer
Social determinants of health and outcomes for children and adults with. . .
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
behavior, and cognition at 5 years old after
CHD surgery in infancy
Poverty
—Lower SES significantly associated with
more perceived cognitive problems in parent
proxy-report model
CHD patients (8–18 years old) and
their caregivers at Cincinnati Children’s
Hospital Medical Center completed QoL
assessment
N=246 Adjusted for 9 covariates
102
—Limbers
Low income/parental educational attainment
—Lower parental educational attainment and
household income associated with less
genetic knowledge in parents of children
with CHD
Survey of parents between 2005 and 2010
of children (0–20 years old) in the LVOT
genetics study at Nationwide Children’s
Hospital
N=287 Adjusted for 12 covariates
103
—Fitzgerald-
Butt
Poverty/educational attainment/
transportation
—Lower maternal educational attainment and
lower SES (free lunch at school) associated
with higher odds of not achieving grade-level
proficiency in literacy and math; distance to
hospital associated with higher odds (1.8) of
not achieving grade-level proficiency in
literacy but not math
Data from Arkansas-born children who had
CHD surgery at Arkansas Children’s Hospital at
<1 year old from 1996–2004
N=458 Adjusted for 16 covariates
51
—Mulkey
Poverty
—In multivariate models, lower SES associated
with memory and learning impairments
Data were combined from two single-center
studies of neurodevelopmental outcomes in
critical CHD
N=268 Adjusted for 9 covariates
104
—Cassidy
Poverty/parental educational attainment
—SES and maternal educational attainment
not associated with risk of screening positive
for autism spectrum disorder in multivariable
analyses
Longitudinal study of children with CHD at
CHOP who underwent surgical repair between
1998 and 2003
N=195 Adjusted for 11 covariates
53
—Bean
Jaworski
Poverty
—Lower family income associated with lower
family quality of life
Cross-sectional design study of children (1–3
years old) with CHD or innocent heart murmur
at Children’s Hospital of Eastern Ontario
(CHEO) and McMaster Children’s Hospital
N=154 Adjusted for 12 covariates
54
—Lee
Adult congenital heart disease—19
Low income/uninsured
—Uninsurance and public insurance
associated with higher risk of hospital
admission via the ED
Patients 12–44 years old with CHD selected
from 2000 to 2003 via California Office State
Health Planning and Development hospital
discharge database
N=9017 Adjusted for 19 covariates
56
—Gurvitz
Parental educational attainment
—Low parental educational attainment
associated with significantly lower mean
scores for purpose of life
Survey (sociodemographic and psychological
well-being) of 380 patients from Adult
Congenital Heart Clinic in Calgary,
Alberta, Canada
N=380 Adjusted for 12 covariates
68
—Balon
Low income
—Public insurance associated with death after
ACHD surgery in bivariate analysis
Analysis of Pediatric Health Information
System (PHIS) from 2000 to 2008 to identify
adult congenital heart surgery admissions
N=97,563 Adjusted for 31 covariates
58
—Kim
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
Low income
—Public insurance associated with high
inpatient resource utilization, which was
associated with higher death rates
Analysis of PHIS from 2000–2008 to identify
adult congenital heart surgery admissions
N=97,563 Adjusted for 9 covariates
59
—Kim
Low income/uninsured
—Higher proportions of uninsured and
publicly insured admitted to hospital from ED
vs. those with private insurance
—Publicly insured costs significantly lower
than privately insured
California State Inpatient Databases
2005–2009 used to conduct retrospective
study on inpatient admissions of CHD patients
10–29 years old and all patients of same age
N=1,202,652 No multivariable analysis of ED admissions
Cost analysis: adjusted for 7 covariates
65
—Lu
Poverty/low income
—Low family income and public insurance
associated with high inpatient resource use
Population-based retrospective study via
Nationwide Inpatient Sample 2005–2009
examining ACHD surgical admissions in 3120
hospitals for patients 18–49 years old
N=16,231 Adjusted for 18 covariates
64
—Bhatt
Transportation barrier
—Increased distance of patient’s home to
specialty center associated with performing
ACHD surgery (for those with moderate or
complex CHD) outside an ACHD specialty
center (with 88% having very low surgical
volume)
Retrospective population analysis used
California’sOffice of Statewide Health
Planning and Development’s discharge
database to analyze ACHD cardiac surgery
outcomes (in patients 21–65 years old) in
California from 2000 to 2011
N=4611 Adjusted for 8 covariates
71
—Fernandes
Uninsured
—Uninsured ACHD patients had increased
rates of loss to follow-up and decreased
successful transfer of care from pediatric to
adult congenital cardiology
Single-institution review of patients >18 years
old with CHD seen by pediatric cardiology
from 2002 to 2007 and their follow-up visits
from 2008 to 2011
N=916 Adjusted for 8 covariates
66
—Bohun
Uninsured
—Uninsurance associated with significantly
higher likelihood of being lost to follow-up
Single-center, cross-sectional study of patients
with CHD who had outpatient visits with
pediatric cardiology before 18 years old
N=306 Adjusted for 9 covariates
67
—Goossens
Poverty
—Income ≤$30,000 associated with poorer
physical quality of life
Cross-sectional study of young adult survivors
of CHD—survey of patients from May 2012 to
December 2013 to examine quality of life
N=218 Adjusted for 23 covariates
60
—Jackson
Uninsured
—Uninsured patients had lower rates of
hospital admission from ED, vs. insured
NEDS (nationwide emergency department
sample) database (includes 30 states) review
to evaluate trend in ED visits among patients
with ACHD from 2006 to 2012
N=72,090 Adjusted for 15 covariates
105
—Agarwal
Low income
—Median annual income <$40,000 and public
insurance associated with increased odds of
readmission after adult congenital heart
surgery
Retrospective cohort study using State
Inpatient Databases for Washington, New
York, Florida, and California from 2009 to 2011
N=9863 Adjusted for 11 covariates
57
—Kim
Poverty
—Lower parental SES associated with poorer
performance on neurocognitive tests
Assessment of neurocognitive function in
patients ≥18 years old born with dTGA
between 1984 and 1995 (n=67) and matched
control group of healthy individuals (n=43)
N=110 Adjusted for 22 covariates
61
——Kasmi
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Table 2. continued
Social determinant(s) of health (SDH) Study design Sample size Notes Reference
number—
first author
Low income
—Non-significant trend toward public
insurance associated with less successful
transfer from pediatric to adult care for
patients with CHD
Single-center retrospective analysis of patients
seen in a dedicated young adult CHD
transition clinic from January 2012 to
December 2015
N=73 Adjusted for 7 covariates
106
—Vaikunth
Low income/uninsured
—Low SES and being uninsured associated
with increased odds of endocarditis-related
admissions
Review of National Inpatient Sample (NIS)
admission in TOF patients (>18 years old),
2000–2014, examining factors associated with
endocarditis admissions
N=18,353 Adjusted for 26 covariates
62
—Egbe
Low income
—Median income ≤$50,000 and public
insurance significantly associated with higher
rate of post-surgical complications vs. private
insurance
ACHD surgery admissions for 18–49 year-olds
from the 2005–2009 Nationwide Inpatient
Sample database
N=16,841 Adjusted for 27 covariates
63
—Setton
Poverty/low income/transportation barriers
—Lower median household income, public
insurance, having PCP, being Philadelphia
resident, history of no-show visits, and shorter
driving distance significantly associated with
clinic non-attendance in bivariable, but not
multivariable, analysis
Analysis of patients (≥18 years old) scheduled
for ACHD outpatient clinic appointment in
Philadelphia and relationship to adverse
events over a 3.5-year period
N=527 Appears to have adjusted for 16 covariates
but does not state which ones were included
in multivariable analysis
72
—Awh
Educational attainment
—Lower educational attainment associated
with decreased exercise frequency
Single-center, cross-sectional study of adults
≥18 years old with CHD in the Washington
Adult Congenital Heart Program at Children’s
National Health System seen during
September 2015–December 2016 to evaluate
factors associated with exercise frequency
N=446 Adjusted for 10 covariates
69
—Connor
Poverty/uninsured/transportation barriers
—ACHD patients with uninsurance, poverty,
and lower educational attainment significantly
more likely to reside farther away from
ACHD center
Geographic information system used to
compare sociodemographic characteristics of
US residents based on their drive times to an
ACHD center
N=56 Adjusted for 5 covariates
70
—Salciccioli
NEDS nationwide emergency department sample.
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stay and higher resource utilization in patients who underwent
congenital heart surgery,
39,40
and lower school functioning and
QOL.
41
One study found that lower maternal educational attainment
was associated with a lower Mental Developmental Index score in
children who underwent the Norwood procedure,
42
but another
study found that maternal educational attainment was not
associated with neurodevelopmental outcomes in multivariable
analyses.
43
Two studies found that distance to the hospital was
actually associated with a lower risk of readmission post-
operatively.
38,44
One study also found no association of uninsur-
ance with post-surgical outcomes.
44
Access to care, loss to follow-up, and hospital readmissions
Nine articles examined the association of SDH with access to
care, loss to follow-up, and hospital admissions (Table 2).
Poverty/low SES, transportation barriers, parental educational
attainment, and immigrant status were significantly associated
with these outcomes. Eight studies documented significant
associations of poverty/low SES with these outcomes, including
increased risk of missed appointments, loss to follow-up, and
hospital readmissions, as well as increases over time of the
proportion of admissions to and bed days in pediatric cardiology
specialty-care centers. For example, a study of 1034 patients in a
large urban pediatric hospital in the Midwest revealed that
Medicaid coverage was associated with a significant higher
adjusted odds of missing at least one scheduled annual
cardiology clinic follow-up visit.
45
A study of nearly 800 patients
showed that Medicaid coverage and lower median household
income were associated with double the unadjusted odds of
missed appointments for cardiac magnetic resonance ima-
ging.
46
Amatchedcase–control study on risk factors for loss
to cardiology clinic follow-up among children and young adults
with CHD documented a 1.2 times greater odds of loss to follow-
up for every $10,000 reduction in family income.
47
Two studies examined transportation barriers and found having
to travel ≥200 miles was associated with missed appointments
(significantly in bivariate analysis, but with a non-significant trend
in multivariable analysis),
48
and residence in rural poor commu-
nities was associated with the longest mean drive time (69 min) to
cardiology clinics.
49
One study of a pediatric cardiology outreach
clinic for immigrant and refugees found a no-show rate that was
higher than the national benchmark.
50
Neurodevelopmental outcomes and QOL
Eight articles examined the association of SDH with neurode-
velopmental outcomes and QOL in children with CHD and
their parents (Table 2). Poverty, parental educational attainment,
and transportation barriers were significantly associated
with worse neurodevelopmental outcomes and QOL in most
studies. Five articles found that poverty/low income was
significantly associated with adverse neurodevelopmental or
QOL outcomes, including decreased intelligence quotient (IQ),
socialization, adaptive behavior, cognition, parental perceived
cognitive problems, genetic knowledge, grade-level literacy and
math proficiency, memory, and family QOL. For example, an
analysis of Arkansas data on children who had CHD surgery at
<1 year old found that poverty was associated with double the
adjusted odds of not achieving grade-level proficiency in
literacy and triple the adjusted odds of not achieving grade-
level proficiency in math.
51
Lower maternal educational attainment was significantly
associated with lower child performance IQ, socialization, adaptive
behavior, and cognition in one study
52
and with lower grade-level
proficiency in literacy in another study.
51
Two studies, however,
found no association of maternal educational attainment with
grade-level proficiency in math or with screening positive on a
measure of autism spectrum disorder.
51,53
A recent study of 140 parents of young children found that,
even when accounting for the severity of the child’s CHD defect
(ranging from an innocent murmur to CHD treatment necessitat-
ing cardiopulmonary bypass), low income was associated with a
significantly lower family QOL.
54
Another study found that SES was
not associated with parental stress.
55
One study showed that a greater travel distance to the hospital
was associated with double the adjusted odds of not achieving
grade-level proficiency in literacy, but no such association was
found for math proficiency.
51
Adult congenital heart disease (ACHD)
Nineteen articles examined associations of SDH with ACHD
outcomes. Poverty/low income (13 studies), uninsurance (5 stu-
dies), educational attainment (3 studies), and transportation
barriers (3 studies) were significantly associated with adverse
ACHD outcomes (Table 2). Poverty/low income was significantly
associated with a variety of adverse ACHD outcomes, including
hospital admissions,
56
hospital readmission
57
and death after
ACHD surgery,
58
higher inpatient resource utilization,
59
physical
QOL,
60
worse neurocognitive test performance,
61
endocarditis-
related hospitalizations,
62
surgical complications,
63
and missed
clinic appointments.
64
For example, analyses of national
databases documented double the odds of inpatient death for
low-income (Medicaid) patients after ACHD surgery
58
and that
patients in the lowest income quartile had significantly higher
adjusted odds of hospitalization for infective endocarditis vs. the
next income quartile.
62
All five studies on uninsurance found significant associations
with adverse ACHD outcomes, including significantly greater odds
of hospitalization,
56,65
outpatient loss to follow-up,
66,67
unsuccess-
ful transfer of care from pediatric to adult congenital cardiology
care,
66
and hospitalization for infective endocarditis.
62
For
example, one study found that uninsured ACHD patients were
significantly less likely to have their pediatric care transferred to
ACHD cardiologists, at only 8%, and most likely to have no follow-
up, at 74%.
66
Three studies found that ACHD patient educational attainment
was significantly associated with adverse ACHD outcomes,
including lower purpose-of-life scores,
68
decreased exercise
frequency,
69
and residing farther from an ACHD center.
70
Three
studies also examined the association of transportation barriers
with adverse ACHD outcomes. One found that transportation
barriers were significantly associated with performance of ACHD
surgery outside of an ACHD specialty center.
71
Another study
revealed that uninsurance, poverty, and lower educational
attainment were significantly associated with ACHD patients with
>6-h drive to the nearest ACHD center.
70
The third study, however,
found no association of driving distance with attendance at ACHD
outpatient clinic appointments.
72
DISCUSSION
This systematic review documented that a wide variety of SDH are
significantly associated with adverse outcomes across the lifespan
of CHD patients, from prenatal diagnosis to ACHD. Indeed, the
study findings dramatically underscore that SDH are significantly
associated with many of the most important and serious CHD
outcomes, including a lower likelihood of prenatal diagnosis,
increased CHD incidence, higher infant mortality, worse post-
surgical outcomes, greater inpatient resource utilization, more
missed clinic appointments, increased loss to follow-up, lower
performance IQ, worse cognition, decreased grade-level profi-
ciency in literacy and math, reduced family QOL, a higher risk for
ACHD endocarditis, more ACHD hospitalizations and hospital
readmissions, unsuccessful transfer of care from pediatric to adult
congenital cardiology care, and increased odds of complications
and death after ACHD surgery.
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These study findings indicate that an urgent priority and one of
the most important interventions for CHD patients would be
routinely screening for SDH, with referrals to appropriate services
for those who screen positive. The study results suggest that this
SDH screening and referral should occur in all CHD care settings,
including prenatal visits, neonatal intensive care units and
pediatric intensive care units, primary-care and specialty practices,
and ACHD clinics. Major national organizations, including the
American Academy of Pediatrics, American College of Cardiology,
American Academy of Family Physicians, and National Academy of
Sciences, Engineering, and Medicine, have all endorsed SDH
screening and referral to appropriate services.
6,73–76
Research
shows that patients and caregivers are comfortable with SDH
screening.
73,77–80
A recent study showed that SDH screening
and referral can reduce the number of SDH and improve child
health.
7
Parent mentors are an evidence-based intervention that has the
potential to prove effective in both reducing SDH and improving
outcomes for children with CHD and their families. Parent mentors
are a special category of community health workers who already
have a child with a particular condition (such as CHD) who then
receive training to help other parents with children with that
condition, including obtaining appropriate healthcare and addres-
sing SDH. A randomized, controlled trial (RCT) of the effects of
parent mentors on children with asthma and their families
revealed that parent mentors were associated with significant
reductions in wheezing, asthma exacerbations, emergency-
department visits, and missed parental work days, while improv-
ing parental self-efficacy, and saving money.
81
Another RCT of a
parent-mentor intervention to enroll uninsured children docu-
mented that parent mentors are significantly more effective than
traditional Medicaid/CHIP outreach and enrollment methods in
insuring uninsured minority children; obtaining insurance faster;
renewing coverage; improving access to primary, dental, and
specialty care; reducing unmet needs and out-of-pocket costs;
achieving parental satisfaction and care quality; and sustaining
long-term coverage; they also saved $6045 per insured child per
year, an 850% return on investment.
82
This RCT resulted in federal
legislation in the 2018 CHIP Reauthorization bill
83
and $120 million
in Centers for Medicare and Medicaid Services funding for parent
mentors.
84–86
Thus, parent mentors could analogously prove to be
highly effective in addressing SDH in children with CHD and their
families.
Study findings on the associations of SDH with ACHD have
important implications for practice, research, and policy. CHD
has morphed from a critical diseaseamongchildrentoachronic
condition in which the number of ACHD patients (~1.3 million)
now exceeds the number children with CHD.
9,58,66
Given that at
least 85% of children with CHD survive to adulthood, there is an
urgent need to provide high-quality specialty care to the
growing ACHD population.
68
Over time, the number of ACHD
hospitalizations has doubled, from ~36,000 in 1998 to >72,000 in
2005.
58
Furthermore, the increasing complexity of ACHD has
warranted creation of an ACHD subspecialty for centers treating
ACHD. SDH screening and appropriate referral to services is thus
increasingly critical for ACHD patients. The study results also
underscore the importance of consistently considering SES as
well as SDH in general when examining health and healthcare
outcomes for fetuses, children, and adults with CHD. Further-
more, the study findings suggest that additional research is
warrantedontheassociationbetweenSDHandCHDinother
developed countries and in developing nations, as well as
country comparative studies, particularly regarding the impact
of variations in welfare state configurations. Until such research
is conducted, caution should be exercised regarding general-
izing our study results beyond populations in the US and
Canada.
This systematic review revealed several unanswered questions.
No published studies were identified on the association of
housing instability with CHD outcomes, and a paucity of research
was noted on several SDH, including food insecurity, transporta-
tion barriers, and lack of health insurance, so more research is
need on these topics. The fewest number of studies was noted for
fetal diagnosis of CHD, so more investigations are needed of which
specific SDH are associated with CHD fetal diagnosis and that
provide a deeper exploration for the reasons behind these
associations. Although several studies found associations of low
maternal educational attainment with infant mortality and other
CHD outcomes, only a single study examined paternal educational
attainment, so an ongoing unanswered question is whether and
how low paternal educational attainment is associated with CHD
outcomes.
Based on the findings of this systematic review, a research
agenda is proposed. More studies are needed on the unan-
swered questions noted above. Research is needed on whether
multiple SDH are associated with even worse CHD outcomes
and how the various SDH might interact. For example, would an
uninsured child with household poverty, food insufficiency, and
low parental educational attainment be at especially high risk
for adverse CHD outcomes? Studies are needed on whether SDH
screening and referral to appropriate services results in
reduction of SDH and improved outcomes. RCTs are urgently
needed of innovative interventions, such as parent mentors, that
might eliminate SDH and achieve better outcomes for children
and adults with CHDs and their families. More research also is
warranted on interventions tailored to reducing SDH for ACHD
patients.
CONCLUSION
SDH are significantly associated with adverse outcomes across the
lifespan of CHD patients, from prenatal diagnosis to ACHD. The
study findings dramatically underscore that SDH are significantly
associated with many of the most important and serious CHD
outcomes, including a lower likelihood of prenatal diagnosis,
increased CHD incidence, higher infant mortality, worse post-
surgical outcomes, greater inpatient resource utilization, more
missed clinic appointments, increased loss to follow-up, lower
performance IQ, worse cognition, decreased grade-level profi-
ciency in literacy and math, reduced family QOL, a higher risk for
ACHD endocarditis, more ACHD hospitalizations and hospital
readmissions, unsuccessful transfer of care from pediatric to adult
congenital cardiology care, and increased odds of complications
and death after ACHD surgery. SDH screening and referral to
appropriate services has the potential to improve outcomes for
CHD patients across the lifespan. RCTs are urgently needed of
innovative interventions, such as parent mentors, that might
eliminate SDH and achieve better outcomes for children and
adults with CHDs and their families.
ACKNOWLEDGEMENTS
We thank Brenda Labbe for her administrative support. No extramural financial
assistance was received in support of this study.
AUTHOR CONTRIBUTIONS
All authors made substantial contributions to the study conception and design,
acquisition of data, analysis, interpretation of data, drafting the article, and revising
the article critically for important intellectual content. Conceptualization: B.D. and
G.F.; methodology: B.D., M.G., and G.F.; data curation: B.D., J.H.L., and M.G.; writing: all
authors.
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ADDITIONAL INFORMATION
The online version of this article (https://doi.org/10.1038/s41390-020-01196-6)
contains supplementary material, which is available to authorized users.
Competing interests: The authors declare no competing interests.
Patient consent: As this was a systematic review, patient consent was not required.
Publisher’s note Springer Nature remains neutral with regard to jurisdictional claims
in published maps and institutional affiliations.
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