1. The Burden of Prenatal Exposure to Alcohol: Quality of Life and Costs Dr. Brenda Stade, RN St. Michael’s Hospital, Toronto

2. Acknowledgement Dr. Bonnie Stevens Dr. Bonnie Stevens Dr. Wendy Ungar Dr. Wendy Ungar Dr. Joseph Beyene Dr. Joseph Beyene Dr. Gideon Koren Dr. Gideon Koren

3. Outline Background Background Purpose Purpose Research Questions Research Questions Significance Significance Methods: Primary Research Questions Methods: Primary Research Questions Results: Primary Research Questions Results: Primary Research Questions Conclusion Conclusion Implications Implications

4. Background In Canada the incidence of Fetal Alcohol Spectrum Disorder (FASD) has been estimated to be 1 to 6 in 1000 live births In Canada the incidence of Fetal Alcohol Spectrum Disorder (FASD) has been estimated to be 1 to 6 in 1000 live births FASD is the leading cause of developmental and cognitive disabilities among Canadian children. FASD is the leading cause of developmental and cognitive disabilities among Canadian children.

5. Relevant Research There are no studies that measure the quality of life of children with FASD. There are no studies that measure the quality of life of children with FASD. Four previous estimates of costs ranged from $74.6 million to $9.69 billion dollars annually to the USA. Four previous estimates of costs ranged from $74.6 million to $9.69 billion dollars annually to the USA.

6. Relevant Research  Previous estimates of costs were limited to FAS and did not reflect other alcohol-related effects.  3 of the 4 studies did not include costs to family/patient.  No research included costs of externalizing behaviors.  All studies were US-based, and estimated annual costs to the nation.

7. Purpose To measure the impact that FAS or FAE has on the Health Related Quality of Life of Canadian children, 8 to 21 years. To measure the impact that FAS or FAE has on the Health Related Quality of Life of Canadian children, 8 to 21 years. To estimate the total costs associated with FAS and FAE at the individual level. To estimate the total costs associated with FAS and FAE at the individual level.

8. Primary Research Questions  What is the Health Related Quality of Life (HRQL) of children with FAS, and FAE?  What are the average annual direct costs and productivity losses per case of FAS and FAE from 1 to 21 years of age in Canada?

9. Secondary Research Questions  What are the factors that influence the costs of FAS and FAE?  What are the lifetime case-specific costs associated with FAS and FAE?  What are the children’s experiences of living day-to day with FAS or FAE?

10. Significance The study provides a major contribution to new knowledge. The study provides a major contribution to new knowledge. It is anticipated that articulating the consequences of prenatal exposure to alcohol may help children with FASD across Canada. It is anticipated that articulating the consequences of prenatal exposure to alcohol may help children with FASD across Canada.

11. Methods Conceptual Perspectives Quality of Life Quality of Life Physical status and functional abilities Physical status and functional abilities 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.

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

13. Methods Conceptual Perspectives Cost Analysis Cost Analysis Analytic perspective was that of the Canadian society. All costs incurred by parents or caregivers were assigned to the child as the unit of analysis. Analytic perspective was that of the Canadian society. All costs incurred by parents or caregivers were assigned to the child as the unit of analysis. Human capital approach was used to measure productivity losses. Human capital approach was used to measure productivity losses.

14. Methods Setting and Study Design Setting Setting Urban and rural communities throughout Canada. Urban and rural communities throughout Canada. Study Design Study Design Multiple cohort cross-sectional. Multiple cohort cross-sectional.

15. Sample HRQL HRQL Children ages 8 to 21 years with FAS or FAE. Children ages 8 to 21 years with FAS or FAE. COST COST Parents of children with FAS and FAE ages 1 to 21 years. Parents of children with FAS and FAE ages 1 to 21 years.

16. Inclusion Criteria : Children 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 enough to complete the questionnaire. Able to understand English well enough to complete the questionnaire.

17. Inclusion Criteria : Parents Parents (biological, adoptive, or foster) of one or more children diagnosed with FASD. Parents (biological, adoptive, or foster) of one or more children diagnosed with FASD. Living with the child who has FASD, or responsible for the care and welfare of that child. Living with the child who has FASD, or responsible for the care and welfare of that child. Able to understand English well enough to complete the questionnaire. Able to understand English well enough to complete the questionnaire.

18. Sample Size Calculation It was determined that a reduction of 0.06 in the quality of life utility score is clinically significant. It was determined that a reduction of 0.06 in the quality of life utility score is clinically significant. SD of quality of life scores in a similar population of children was 0.21. SD of quality of life scores in a similar population of children was 0.21. Assuming an α of 0.05 and ß of 0.20 and a 2 tailed test, it was necessary to interview 99 participants. Assuming an α of 0.05 and ß of 0.20 and a 2 tailed test, it was necessary to interview 99 participants.

19. Data Collection HRQL The Health Utilities Index Mark 3 (HUI3) The Health Utilities Index Mark 3 (HUI3) A multi-attribute health status classification system. A multi-attribute health status classification system. A multi-attribute utility function. A multi-attribute utility function.

20. Data Collection: HUI3 The health status classification system (questionnaire) is comprised of 33 questions that measures 8 health attributes. The health status classification system (questionnaire) is comprised of 33 questions that measures 8 health attributes.

21. Data Collection: HUI3 Vision Vision Hearing Hearing Speech Speech Ambulation Ambulation Dexterity Dexterity Emotion Emotion Cognition Cognition Pain Pain

22. Data Collection HUI3 VISION VISION 1.Are you able to see well enough to read ordinary newsprint without glasses or contact lenses? __Yes __No __Don’t Know __Refused 1.Are you able to see well enough to read ordinary newsprint without glasses or contact lenses? __Yes __No __Don’t Know __Refused

23. Data Collection: HUI3 The unique combination of responses of each set of questions in the HUI3 determines the level of the health attribute. The unique combination of responses of each set of questions in the HUI3 determines the level of the health attribute. Each attribute has five to six defined levels ranging from normal function to severe dysfunction. Each attribute has five to six defined levels ranging from normal function to severe dysfunction.

24. Attribute Levels: Vision 1. 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. 3. Unable to recognize a friend … even with glasses. 4. Unable to read newsprint even with glasses. 5. Unable to read newsprint or recognize a friend …, even with glasses. 6.Unable to see at all.

25. Health Services Utilization Inventory (HSUI) Health Services Utilization Inventory (HSUI) Twenty-five page inventory. Twenty-five page inventory. Direct costs – medical, education, social services, out-of-pocket costs to parents. Direct costs – medical, education, social services, out-of-pocket costs to parents. Productivity losses. Productivity losses. Data Collection Costs

26. If speech therapist selected: If speech therapist selected: How many visits did _________(name of child) have in the last 3 months? _______ How many visits did _________(name of child) have in the last 3 months? _______ Where did this health visit take place? Private Office__________ Hospital Clinic_________ Emergency Room_________ Other _________________(please specify) Data Collection: HSUI

27. Data Analysis: HRQL Health status of each child was described by a eight-element vector (x 1, x 2...and x 8 ), in which x i represents the level (1 to 5, or 1 to 6) of the attribute i. Health status of each child was described by a eight-element vector (x 1, x 2...and x 8 ), in which x i represents the level (1 to 5, or 1 to 6) of the attribute i. A utility equation was applied to the multi-attribute health state description of each participant. A utility equation was applied to the multi-attribute health state description of each participant. Utilities were used to measure a single cardinal value between 0.0 and 1.0 (0 = death; 1 = perfect health) to reflect a HRQL score. Utilities were used to measure a single cardinal value between 0.0 and 1.0 (0 = death; 1 = perfect health) to reflect a HRQL score.

29. DATA ANALYSIS: CALCULATING HRQL SCORES X1b1 X1b1 1.001 1.001 0.982 0.982 0.893 0.893 0.844 0.844 0.755 0.755 0.616 0.616 u = 1.371 (0.75 x b2 x b3 x b4 x b5 x b6 x b7 x b8) - 0.371 u = 1.371 (0.75 x b2 x b3 x b4 x b5 x b6 x b7 x b8) - 0.371

30. Data Analysis HRQL  Compare HRQL scores of children with FAS/FAE to a reference group (One sample t-test).  Compare HRQL scores of children with FAS to those with FAE (t-test for independent groups).  Correlate the child versus parent reports of HRQL (Pearson Correlation Coefficient).

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

32. HRQL Results: Participants (n=126) SEX Female54 (43 %) Male72 (57 %) Male72 (57 %) AGE AGE 8-1248 (38 %) 8-1248 (38 %) 13-1740 (32 %) 13-1740 (32 %) 18-2138 (30%) 18-2138 (30%) Age Mean: 14.5 years DIAGNOSIS FAS 56 (44 %) FAE 70 (56 %) RELATIONSHIP Biological 14(11 %) Adoptive 70 (56 %) Foster 42(33 %) CULTURAL GROUP Native 57 (45 %) Euro-Canadian 69 (55 %)

33. Participants by Geographical Regions

34. RESULTS: MEAN HRQL SCORES MEAN MEAN 0.47 0.47RANGE -0.22 to 0.96 SD0.27

35. MEAN SINGLE ATTRIBUTE UTILITY SCORES AttributeFAS/FAE AttributeFAS/FAE Vision1.00 Vision1.00 Hearing 0.99 Hearing 0.99 Speech 0.97 Speech 0.97 Ambulation 1.00 Dexterity1.00 Ambulation 1.00 Dexterity1.00 Emotion0.76 Emotion0.76 Cognition0.83 Cognition0.83 Pain1.00 Pain1.00

36. Mean HRQL FAS/FAE versus Reference Group N Min Min Max MaxMean SD SDFAS/FAE 126-0.220.96 0.47 0.47 0.27 0.27 Reference4423 0.91 0.910.95 0.93 0.93 0.03 0.03

37. MEAN HQRL: FAS versus FAE NMeanSDCI FAS560.440.27 0.37, 0.52 FAE700.500.27 0.44, 0.57

38. Child versus Parent Reports r = 0.932 r = 0.932

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

40. Components of Direct Costs

41. Contributors of Cost

42. Adjusted Cost Severity of the child’s condition, age of the child, and geographical setting significantly impacted on costs. Severity of the child’s condition, age of the child, and geographical setting significantly impacted on costs. The summary adjusted value of annual costs was $14,342 (95% CI, $12,986; $15,698.). The summary adjusted value of annual costs was $14,342 (95% CI, $12,986; $15,698.).

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

44. Total Life-Time Costs Age Group Cost Item Average Annual Cost Present value of costs discounted at 3% to age 0 Infancy 0 to 1 year hospitalization$15,976 $ 15,511 1 to 21 years all$14,342$214,643 21 to 65 years direct health care productivity losses institutionalization $ 5,683 $29,736$11,160 $ 74,902 $391,921$147,089 Present value of total lifetime costs discounted back to age 0 $844,066.

45. Conclusions Burden of prenatal exposure to alcohol is profound. Burden of prenatal exposure to alcohol is profound. First of such research on the topic of FAS. First of such research on the topic of FAS. Anticipated by articulating the burden of prenatal exposure to alcohol, that this study will help children with FASD across Canada. Anticipated by articulating the burden of prenatal exposure to alcohol, that this study will help children with FASD across Canada.

46. Implications for Practice and Policy Less emphasis on behaviors and more on the emotional health of these children. Less emphasis on behaviors and more on the emotional health of these children. Early diagnostic programs. Early diagnostic programs. Health and educational programs to build self-esteem and success. Health and educational programs to build self-esteem and success. Programs to deal with anxiety and depression. Programs to deal with anxiety and depression.

47. Implications for Practice and Policy Emphasis on prevention strategies. Emphasis on prevention strategies. Benefit of a particular prevention policy can be calculated. Benefit of a particular prevention policy can be calculated. Decision-makers should be aware of the substantial long-term economic impact on parents. Decision-makers should be aware of the substantial long-term economic impact on parents.

48. Implications for Research Evaluation of treatment and educational programs on HRQL. Evaluation of treatment and educational programs on HRQL. Impact of child-parent relationship on HRQL. Impact of child-parent relationship on HRQL. Economic evaluations of primary and secondary prevention strategies. Economic evaluations of primary and secondary prevention strategies.

49. Implications for Research Future studies that include prospective data of costs in infancy. Future studies that include prospective data of costs in infancy. Methods used in the cost section could benefit from further research. Methods used in the cost section could benefit from further research. More qualitative research. More qualitative research. Development of a quality of life tool specific for children with FAS and FAE. Development of a quality of life tool specific for children with FAS and FAE.

50. Acknowledgement Research Training Grant, Hospital for Sick Children. Research Training Grant, Hospital for Sick Children. Health Utilities Inc. for use of their tool Health Utilities Inc. for use of their tool Valerie Fine Award, Mount Sinai Hospital. Valerie Fine Award, Mount Sinai Hospital. Department of Pediatrics, St. Michael’s Hospital. Department of Pediatrics, St. Michael’s Hospital.