The Burden of Prenatal The Burden of Prenatal Exposure to Alcohol: Exposure to Alcohol:

Quality of Life and CostsQuality of Life and Costs

Dr. Brenda Stade, RN Dr. Brenda Stade, RN St. Michael’s Hospital, TorontoSt. Michael’s Hospital, Toronto

AcknowledgementAcknowledgement Dr. Bonnie StevensDr. Bonnie Stevens

Dr. Wendy UngarDr. Wendy Ungar

Dr. Joseph BeyeneDr. Joseph Beyene

Dr. Gideon KorenDr. Gideon Koren

OutlineOutline BackgroundBackground PurposePurpose Research QuestionsResearch Questions SignificanceSignificance Methods: Primary Methods: Primary

Research QuestionsResearch Questions Results: Primary Results: Primary

Research QuestionsResearch Questions ConclusionConclusion ImplicationsImplications

BackgroundBackground

In Canada the incidence of Fetal Alcohol In Canada the incidence of Fetal Alcohol Spectrum Disorder (FASD) has been Spectrum Disorder (FASD) has been estimated to be 1 to 6 in 1000 live estimated to be 1 to 6 in 1000 live birthsbirths

FASD is the leading cause of FASD is the leading cause of developmental and cognitive disabilities developmental and cognitive disabilities among Canadian children. among Canadian children.

Relevant ResearchRelevant Research

There are no studies that measure the There are no studies that measure the quality of life of children with FASD.quality of life of children with FASD.

Four previous estimates of costs ranged Four previous estimates of costs ranged from $74.6 million to $9.69 billion from $74.6 million to $9.69 billion dollars annually to the USA.dollars annually to the USA.

Relevant ResearchRelevant Research Previous estimates of costs were Previous estimates of costs were

limited to FAS and did not reflect limited to FAS and did not reflect other alcohol-related effects.other alcohol-related effects.

3 of the 4 studies did not include 3 of the 4 studies did not include costs to family/patient.costs to family/patient.

No research included costs of No research included costs of externalizing behaviors.externalizing behaviors.

All studies were US-based, and All studies were US-based, and estimated annual costs to the nation.estimated annual costs to the nation.

PurposePurpose

To measure the impact that FAS or To measure the impact that FAS or FAE has on the Health Related FAE has on the Health Related Quality of Life of Canadian children, Quality of Life of Canadian children, 8 to 21 years.8 to 21 years.

To estimate the total costs To estimate the total costs associated with FAS and FAE at the associated with FAS and FAE at the individual level.individual level.

Primary Research Primary Research QuestionsQuestions

What is the Health Related What is the Health Related Quality of Life (HRQL) of children Quality of Life (HRQL) of children with FAS, and FAE?with FAS, and FAE?

What are the average annual What are the average annual direct costs and productivity direct costs and productivity losses per case of FAS and FAE losses per case of FAS and FAE from 1 to 21 years of age in from 1 to 21 years of age in Canada?Canada?

Secondary Research Secondary Research QuestionsQuestions

What are the factors that influence What are the factors that influence the costs of FAS and FAE?the costs of FAS and FAE?

What are the lifetime case-specific What are the lifetime case-specific costs associated with FAS and FAE?costs associated with FAS and FAE?

What are the children’s experiences What are the children’s experiences of living day-to day with FAS or FAE?of living day-to day with FAS or FAE?

SignificanceSignificance

The study provides a major The study provides a major contribution to new knowledge.contribution to new knowledge.

It is anticipated that It is anticipated that articulating the consequences articulating the consequences of prenatal exposure to alcohol of prenatal exposure to alcohol may help children with FASD may help children with FASD across Canada.across Canada.

MethodsMethodsConceptual Conceptual

PerspectivesPerspectives Quality of Life Quality of Life

Physical status and functional Physical status and functional abilitiesabilities

Psychological and well-being Psychological and well-being Social interactions Social interactions Economic and/or vocational status Economic and/or vocational status Spiritual/religious status.Spiritual/religious status.

MethodsMethodsConceptual Conceptual

PerspectivesPerspectives Quality of LifeQuality of Life

Health-related quality of life Health-related quality of life (HRQL) has been used to describe (HRQL) has been used to describe the subset of QOL directly related the subset of QOL directly related to an individual’s health.to an individual’s health.

MethodsMethodsConceptual Conceptual

PerspectivesPerspectives Cost AnalysisCost Analysis Analytic perspective was that of the Analytic perspective was that of the

Canadian society. All costs incurred Canadian society. All costs incurred by parents or caregivers were by parents or caregivers were assigned to the child as the unit of assigned to the child as the unit of analysis.analysis.

Human capital approach was used to Human capital approach was used to measure productivity losses.measure productivity losses.

MethodsMethodsSetting and Study Setting and Study

DesignDesign

SettingSetting Urban and rural communities Urban and rural communities

throughout Canada.throughout Canada. Study DesignStudy Design

Multiple cohort cross-sectional.Multiple cohort cross-sectional.

SampleSample

HRQLHRQL Children ages 8 to 21 years Children ages 8 to 21 years

with FAS or FAE. with FAS or FAE.

COSTCOST Parents of children with FAS Parents of children with FAS

and FAE ages 1 to 21 years.and FAE ages 1 to 21 years.

Inclusion Criteria : Inclusion Criteria : ChildrenChildren

Diagnosis of FASD.Diagnosis of FASD.

Eight (8) to 21 years of age.Eight (8) to 21 years of age.

Able to understand English well Able to understand English well enough to complete the enough to complete the questionnaire.questionnaire.

Inclusion Criteria : Inclusion Criteria : ParentsParents

Parents (biological, adoptive, or Parents (biological, adoptive, or foster) of one or more children foster) of one or more children diagnosed with FASD. diagnosed with FASD.

Living with the child who has Living with the child who has FASD, or responsible for the care FASD, or responsible for the care and welfare of that child.and welfare of that child.

Able to understand English well Able to understand English well enough to complete the enough to complete the questionnaire. questionnaire.

Sample Size Sample Size CalculationCalculation

It was determined that a reduction It was determined that a reduction of 0.06 in the quality of life utility of 0.06 in the quality of life utility score is clinically significant. score is clinically significant.

SD of quality of life scores in a SD of quality of life scores in a similar population of children was similar population of children was 0.21. 0.21.

Assuming an Assuming an αα of 0.05 and ß of 0.20 of 0.05 and ß of 0.20 and a 2 tailed test, it was necessary and a 2 tailed test, it was necessary to interview 99 participants. to interview 99 participants.

Data CollectionData CollectionHRQLHRQL

The Health Utilities Index Mark 3 The Health Utilities Index Mark 3 (HUI3)(HUI3) A multi-attribute health status A multi-attribute health status

classification system. classification system. A multi-attribute utility function.A multi-attribute utility function.

Data Collection: HUI3Data Collection: HUI3

The health status classification The health status classification system (questionnaire) is system (questionnaire) is comprised of 33 questions that comprised of 33 questions that measuresmeasures 8 health attributes.8 health attributes.

Data Collection: HUI3Data Collection: HUI3 VisionVision HearingHearing SpeechSpeech Ambulation Ambulation DexterityDexterity EmotionEmotion CognitionCognition PainPain

Data CollectionData CollectionHUI3HUI3

VISIONVISION 1.1. Are you able to see well enough to Are you able to see well enough to

read read ordinary newsprint without ordinary newsprint without glasses or contact lenses?glasses or contact lenses? ____

YesYes ____ NoNo____ Don’t KnowDon’t Know____ RefusedRefused

Data Collection: HUI3Data Collection: HUI3 The unique combination of responses of The unique combination of responses of

each set of questions in the HUI3 each set of questions in the HUI3 determines the level of the health determines the level of the health attribute.attribute.

Each attribute has five to six defined Each attribute has five to six defined levels ranging from normal function to levels ranging from normal function to severe dysfunction. severe dysfunction.

Attribute Levels: VisionAttribute Levels: Vision1. Able to see well enough to read ordinary

newsprint and recognize a friend on the other side of the street, without glasses or contacts.

2. Able to see well enough … , but with glasses.2. Able to see well enough … , but with glasses.

3. Unable to recognize a friend … even with 3. Unable to recognize a friend … even with glasses.glasses.

4. Unable to read newsprint even with glasses.4. Unable to read newsprint even with glasses.

5. Unable to read newsprint or recognize a friend 5. Unable to read newsprint or recognize a friend … , even with glasses.… , even with glasses.

6.Unable to see at all.

Health Services Utilization Health Services Utilization Inventory (HSUI)Inventory (HSUI)

Twenty-five page inventory.Twenty-five page inventory. Direct costs – medical, education, Direct costs – medical, education,

social services, out-of-pocket costs social services, out-of-pocket costs to parents.to parents.

Productivity losses.Productivity losses.

Data CollectionData CollectionCostsCosts

If speech therapist selected:If speech therapist selected: How many visits did _________(name of How many visits did _________(name of

child) have in the last 3 months? _______child) have in the last 3 months? _______Where did this health visit take place?Where did this health visit take place?Private Office__________Private Office__________Hospital Clinic_________Hospital Clinic_________Emergency Room_________Emergency Room_________Other _________________(please specify)Other _________________(please specify)

Data Collection: HSUIData Collection: HSUI

Data Analysis: HRQLData Analysis: HRQL Health status of each child was described Health status of each child was described

by a eight-element vector (xby a eight-element vector (x11, x, x22...and x...and x88), ), in which xin which xi i represents the level (1 to 5, or represents the level (1 to 5, or 1 to 6) of the attribute 1 to 6) of the attribute i.i.

A utility equation was applied to the A utility equation was applied to the multi-attribute health state description of multi-attribute health state description of each participant. each participant.

Utilities were used to measure a single Utilities were used to measure a single cardinal value between 0.0 and 1.0 (0 cardinal value between 0.0 and 1.0 (0 = death; 1 = perfect health) to reflect a = death; 1 = perfect health) to reflect a HRQL score.HRQL score.

DATA ANALYSIS: DATA ANALYSIS: CALCULATING HRQL SCORESCALCULATING HRQL SCORES

X1X1 b1b1 1.001.00 11 0.980.98 22 0.890.89 33 0.840.84 44 0.750.75 55 0.610.61 66

u =u = 1.3711.371 ((00.75.75 x b2 x b3 x b4 x b5 x x b2 x b3 x b4 x b5 x b6 x b6 x b7 x b8) - 0.371 b7 x b8) - 0.371

Data AnalysisData Analysis HRQL HRQL

Compare HRQL scores of children with FAS/FAE Compare HRQL scores of children with FAS/FAE to a reference group (to a reference group (One sample t-testOne sample t-test).).

Compare HRQL scores of children with FAS to Compare HRQL scores of children with FAS to those with FAE (those with FAE (t-test for independent groupst-test for independent groups).).

Correlate the child versus parent reports of Correlate the child versus parent reports of HRQL (HRQL (Pearson Correlation CoefficientPearson Correlation Coefficient).).

Data AnalysisData Analysis Cost Cost

Average annual total costs were Average annual total costs were calculated at the patient level calculated at the patient level by summing the costs for each by summing the costs for each child in each cost component child in each cost component and dividing by the sample size.and dividing by the sample size.

HRQL Results: HRQL Results: Participants Participants

(n=126)(n=126)SEXSEX FemaleFemale 54 (43 %)54 (43 %)

MaleMale 72 (57 %) 72 (57 %) AGEAGE

8-128-12 48 (38 %)48 (38 %) 13-1713-17 40 (32 40 (32 %)%) 18-2118-21 38 38 (30%) (30%)

Age Mean: 14.5 Age Mean: 14.5 yearsyears

DIAGNOSISDIAGNOSIS FAS FAS 56 (44 %) 56 (44 %)FAEFAE 70 (56 %) 70 (56 %)RELATIONSHIP RELATIONSHIP Biological Biological 14 14 (11 %)(11 %)Adoptive Adoptive 70 (56 %) 70 (56 %)FosterFoster 42 42 (33 %)(33 %)CULTURAL GROUPCULTURAL GROUPNative Native 57 (45 %) 57 (45 %)Euro-Canadian 69 (55 %)Euro-Canadian 69 (55 %)

Participants by Participants by Geographical RegionsGeographical Regions

Central33%n=42

East29%n=36

West38%n=48

RESULTS: RESULTS: MEAN HRQL SCORESMEAN HRQL SCORES

MEANMEAN

0.470.47

RANGERANGE

-0.22 to -0.22 to 0.960.96

SDSD

0.270.27

MEAN SINGLE MEAN SINGLE ATTRIBUTE UTILITY ATTRIBUTE UTILITY

SCORESSCORES AttributeAttribute FAS/FAEFAS/FAE VisionVision 1.001.00 HearingHearing 0.990.99 SpeechSpeech 0.970.97 AmbulationAmbulation 1.00 1.00

DexterityDexterity 1.001.00 EmotionEmotion 0.760.76 CognitionCognition 0.830.83 PainPain 1.001.00

Mean HRQLMean HRQLFAS/FAE versus Reference GroupFAS/FAE versus Reference Group

0.47

0.92

00.10.20.30.40.50.60.70.80.91

FAE/FAE Reference

NN MinMin MaxMax MeaMeann

SDSD

FAS/FAEFAS/FAE

126126 -0.22-0.22 0.960.96

0.470.47

0.270.27

ReferenceReference 44234423 0.910.91 0.950.95 0.930.93 0.030.03

MEAN HQRL: FAS versus MEAN HQRL: FAS versus FAEFAE

NN MeanMean SDSD CICI

FASFAS 5656 0.440.44 0.270.27 0.37, 0.520.37, 0.52

FAEFAE 7070 0.500.50 0.270.27 0.44, 0.570.44, 0.57

0.440.5

0.00

0.50

1.00

FAS FAE

Child versus Parent Child versus Parent ReportsReports

r = 0.932r = 0.932

PARENT

1.0.8.6.4.20.0-.2-.4

CH

ILD

1.0

.8

.6

.4

.2

0.0

-.2

Average Annual Total Average Annual Total CostsCosts

Average annual unadjusted Average annual unadjusted costs costs per case of FAS and per case of FAS and FAE, ages 1 to 21 years, in FAE, ages 1 to 21 years, in Canada = Canada = $$13,109.13,109.

Components of Direct Components of Direct CostsCosts

Medical33%

Out-of-Pocket

8%

Social Services

24%

Education35.5%

Contributors of CostContributors of Cost

Government Ministries

81%

Parent19%

Adjusted CostAdjusted Cost

Severity of the child’s condition, age Severity of the child’s condition, age of the child, and geographical setting of the child, and geographical setting significantly impacted on costs.significantly impacted on costs.

The summary adjusted value of The summary adjusted value of annual costs was $14,342 (95% CI, annual costs was $14,342 (95% CI, $12,986; $15,698.).$12,986; $15,698.).

Cost to the NationCost to the Nation

Cost of FAS/FAE annually to Cost of FAS/FAE annually to Canada of those 1 to 21 years Canada of those 1 to 21 years old, was $344,208,000 (95% CI old, was $344,208,000 (95% CI $311,664,000; $376,752,000).$311,664,000; $376,752,000).

Total Life-Time CostsTotal Life-Time CostsAge GroupAge Group Cost ItemCost Item AverageAverage

Annual Annual CostCost

Present value Present value of costs of costs discounted at discounted at 3% to age 03% to age 0

Infancy Infancy

0 to 1 year0 to 1 year hospitalizationhospitalization $15,976$15,976 $ 15,511$ 15,511

1 to 21 years1 to 21 years allall $14,342$14,342 $214,643$214,643

21 to 65 21 to 65 yearsyears

direct health direct health carecare

productivity productivity losseslosses

institutionalizatiinstitutionalizationon

$ 5,683$ 5,683

$29,736$29,736

$11,160$11,160

$ 74,902$ 74,902

$391,921$391,921

$147,089$147,089

Present Present value of value of total total lifetime lifetime costs costs discounted discounted back to age back to age 00

$844,066.$844,066.

ConclusionsConclusions

Burden of prenatal exposure to alcohol Burden of prenatal exposure to alcohol is profound.is profound.

First of such research on the topic of First of such research on the topic of FAS.FAS.

Anticipated by articulating the burden Anticipated by articulating the burden of prenatal exposure to alcohol, that of prenatal exposure to alcohol, that this study will help children with FASD this study will help children with FASD across Canada.across Canada.

Implications for Implications for Practice and PolicyPractice and Policy

Less emphasis on behaviors and Less emphasis on behaviors and more on the emotional health of more on the emotional health of these children. these children.

Early diagnostic programs.Early diagnostic programs. Health and educational Health and educational

programs to build self-esteem programs to build self-esteem and success.and success.

Programs to deal with anxiety Programs to deal with anxiety and depression.and depression.

Implications for Implications for Practice and PolicyPractice and Policy

Emphasis on prevention strategies.Emphasis on prevention strategies.

Benefit of a particular prevention policy Benefit of a particular prevention policy can be calculated.can be calculated.

Decision-makers should be aware of the Decision-makers should be aware of the substantial long-term economic impact on substantial long-term economic impact on parents.parents.

Implications for Implications for Research Research

Evaluation of treatment and Evaluation of treatment and educational programs on HRQL.educational programs on HRQL.

Impact of child-parent Impact of child-parent relationship on HRQL.relationship on HRQL.

Economic evaluations of primary Economic evaluations of primary and secondary prevention and secondary prevention strategies.strategies.

Implications for Implications for ResearchResearch

Future studies that include prospective Future studies that include prospective data of costs in infancy.data of costs in infancy.

Methods used in the cost section could Methods used in the cost section could benefit from further research.benefit from further research.

More qualitative research.More qualitative research.

Development of a quality of life tool Development of a quality of life tool specific for children with FAS and FAE.specific for children with FAS and FAE.

AcknowledgementAcknowledgement Research Training Research Training

Grant, Hospital for Grant, Hospital for Sick Children.Sick Children.

Health Utilities Inc. for Health Utilities Inc. for use of their tooluse of their tool

Valerie Fine Award, Valerie Fine Award, Mount Sinai Hospital.Mount Sinai Hospital.

Department of Department of Pediatrics, St. Pediatrics, St. Michael’s Hospital.Michael’s Hospital.