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date: 01 October 2020

Burden and Social Cost of Fetal Alcohol Spectrum Disorders

Abstract and Keywords

Damage to the central nervous system is a unifying concept for nearly all of the diagnoses that fall under the Fetal Alcohol Spectrum Disorders (FASD) umbrella. Thus, FASD are an important public health and social problem worldwide that consumes a large amount of resources, both economic and societal by imparting a large burden on society through such sectors as the healthcare system, mental health and substance abuse treatment services, foster care, the criminal justice system, and the long-term care of individuals with intellectual and physical disabilities. Existing estimates of the economic impact of FASD demonstrate significant cost implications on the individual, the family and society. Many of the costs associated with FASD can be reduced with the implementation of effective social policies and intervention programs.

Keywords: burden, cost, fetal alcohol spectrum disorders, fetal alcohol syndrome, prevalence

Introduction

Fetal alcohol syndrome (FAS) was first described by Lemoine and colleagues (Lemoine, Harousseau, Borteyru, & Menuet, 1968) in France. Five years later, Jones, Smith, Ulleland, and Streissguth (1973) described the diagnostic criteria for FAS. Since this time, it has been recognized that individuals prenatally exposed to alcohol exhibit a wide variety of symptoms. As such, the non-diagnostic umbrella term fetal alcohol spectrum disorders (FASD) was coined in an effort to capture the range of effects that can occur in individuals prenatally exposed to alcohol. According to the Canadian FASD Diagnostic Guidelines (Chudley et al., 2005), FASD includes the following three diagnoses: FAS, partial FAS (pFAS), and alcohol-related neurodevelopmental disorder (ARND). However, some diagnostic guidelines (e.g., Stratton, Howe, & Battaglia, 1996) also include alcohol-related birth defects (ARBD) within this spectrum. FAS is the least prevalent form of FASD; however, it is the most severe and visibly identifiable form of FASD. The diagnosis of FAS requires the following three facial anomalies: (i) short palpebral fissure length, (ii) smooth or flattened philtrum, and (iii) thin upper lip (Chudley et al., 2005); and the diagnosis of pFAS requires the simultaneous presentation of two of the facial anomalies. Lastly, ARND involves damage to the central nervous system, although the criteria for the type and extent of damage lack consensus. However, ARND is recognized to be the most prevalent diagnostic category of FASD, representing as many as 80–90% of FASD cases (Chudley, 2008).

Alcohol is a teratogen (an agent that can impair fetal development and cause birth defects) that can affect any organ or system of the fetus. While research has not been able to delineate the pattern, amount, or critical period of prenatal alcohol exposure necessary for structural and/or functional teratogenesis, it is apparent that a dose–response relationship exists (Jacobson & Jacobson, 1994, 1999; Sood et al., 2001). However, in addition to the amount and frequency of alcohol consumption, the range and severity of symptoms are associated with various effect modifiers, including the stage of pregnancy when alcohol is consumed, the overall level of health of the mother, and the genetic makeup of both the mother and fetus.

Epidemiology of FASD

Prevalence

Prevalence refers to the proportion of a population found to have a specific condition, while incidence refers to the occurrence of new cases of a specific condition in a population—both within a specified period of time. In studies of FASD (and congenital malformations in general), prevalence is the epidemiological measure usually employed, as “incidence” refers to the occurrence of these conditions among the susceptible population of embryos (Rothman, Greenland, & Lash, 2012). Given that prenatal alcohol exposure can lead to early embryonic or fetal death, it is difficult to ascertain the population at risk, as cases of FASD at birth and thereafter represent only the fetuses that survived long enough to be recorded as births. Accordingly, the rest of the discussion will refer to measures of prevalence.

One of the first steps towards truly understanding the scope and severity of FASD is to determine the prevalence at which it occurs. Prevalence estimates are not only vital for informing policymakers of the impact of these conditions, but also integral to raising awareness of FASD. It should be noted that the variations in prevalence rates reported do not solely arise from differences in maternal drinking behavior and degrees of public awareness of alcohol’s detrimental effects during pregnancy—they also stem from differences in the diagnostic criteria and assessment techniques used, methods of surveillance, and varying sample/population demographics (e.g., age of participants).

General Populations

Data on the prevalence of FAS and FASD around the world are scarce. The majority of studies reporting on the prevalence of FASD have been conducted in North America (particularly the United States) and Europe. Many well-conducted prevalence studies have also been carried out in South Africa. The most recent studies conducted in the United States have estimated the prevalence of FAS to be 3.0–8.4 per 1,000, and the prevalence of FASD to be 11.1–33.5 per 1,000 (May et al., 2014, 2015). In Croatia, Petković and Barišić (2010, 2013) have reported the prevalence of FAS to be 6.4–17.0 per 1,000 and the prevalence of FASD to be 40.7–66.8 per 1,000. In France, the reported prevalence of FAS and FASD ranges from 1.9–48.5 per 1,000 (de Chazeron et al., 2008; Serreau et al., 2002) and 5.6–66.0 per 1,000 (Serreau et al., 2002; Toutain & Lejeune, 2008), respectively. The prevalence of FAS and FASD in Italy have been reported to be 7.4–8.2 per 1,000 and 40.5–47.1 per 1,000, respectively (May et al., 2006, 2011). Although the above prevalence rates are notably high for a largely preventable disorder, the highest-known prevalence of FAS and FASD has been reported in South Africa: from 46.4–100.0 per 1,000 (May et al., 2000; Olivier, Urban, Chersich, Temmerman, & Viljoen, 2013) and 63.9–207.5 per 1,000 (May et al., 2013; Urban et al., 2015), respectively.

Aboriginal Populations

It is a commonly held belief that alcohol abuse during pregnancy occurs more frequently among Aboriginal women than in the general population, and that, as a result, the prevalence of FASD is higher among this population. However, given that FASD prevalence studies are more likely to take place in communities where there is a concern for a high rate of occurrence, it should be acknowledged that prevalence studies conducted among individual reserves/communities may not be generalizable to the Aboriginal population as a whole.

Based on the current literature in Australia, the prevalence of FAS and FASD among Aboriginal populations have been reported to range from 2.0–9.2 per 1,000 (Fitzpatrick et al., 2015; Harris & Bucens, 2003) and 4.1–120.4 per 1,000 (Fitzpatrick et al., 2015; Mutch, Watkins, & Bower, 2015), respectively. In Canada, the prevalence of FAS and FASD among Aboriginal populations reportedly range from 7.2–120.7 per 1,000 (Robinson et al., 1987; Williams, Odaibo, & McGee, 1999) and 7.0–189.7 per 1,000 (Robinson et al., 1987; Werk, Cui, & Tough, 2013), respectively. Lastly, the documented prevalence of FAS and FASD among Aboriginal populations in the United States range from 2.0–9.3 per 1,000 (Fox et al., 2015; Quaid, Kirkpatrick, Nakamura, & Aase, 1993) and 3.7–18.7 per 1,000 (May, Hymbaugh, Aase, & Samet, 1983; Quaid et al., 1993), respectively.

Children in Care

Children often enter a childcare system (e.g., orphanage, foster care, child welfare system) due to unfavorable circumstances (e.g., maternal alcohol and/or drug problems, and child abuse and neglect). Such circumstances increase the odds of prenatal alcohol exposure, and this population can therefore be regarded as at high risk for FASD. In fact, parental substance abuse has been identified as one of the main reasons for a child’s ending up in an orphanage or foster care (Burd, Cohen, Shaw, & Norris, 2011; Herrick, Hudson, & Burd, 2011; Miller et al., 2007).

It is clear from the current literature that the prevalence of FAS and FASD varies greatly, depending on the childcare setting (Lange, Shield, Rehm, & Popova, 2013). Specifically, the lowest prevalence of FAS among children in childcare settings has been observed among children in the child welfare system in the United States (5.3 per 1,000; Ringeisen, Casanueva, Urato, & Cross, 2008), whereas the highest prevalence of FAS among children in childcare settings has been reported among children residing in an orphanage for children with special needs in Russia (680 per 1,000; Pal’chik & Legon’kova, 2011). Furthermore, the prevalence of FASD among children in childcare settings appears to be the lowest among foster and pre-adoption children in Israel (Tenenbaum et al., 2011) and adoptees from Eastern Europe in the United States, at 40 per 1,000 (Loman, Wiik, Frenn, Pollak, & Gunnar, 2009; Miller, Chan, Tirella, & Perrin, 2009), and the highest among adoptees in Sweden from Eastern Europe, at 521 per 1,000 (Landgren, Landgren, Strömland, & Andersson Grönlund, 2010).

In a recently conducted review and meta-analysis by the authors, it was estimated that the overall pooled prevalence of FAS and FASD among children and youth in the care of a childcare system was 6.0% (60 per 1,000; 95% confidence interval [CI]: 38–85 per 1,000) and 16.9% (169 per 1,000; 95% CI: 109–238 per 1,000), respectively, based on studies that utilized active case ascertainment (ACA; Lange et al., 2013). The prevalence of FASD among children in care has been estimated for a number of countries, and, as indicated above, appears to vary depending on the childcare setting (Lange et al., 2013).

Correctional Population

People with FASD often display characteristics such as hyperactivity, impulsivity, aggressiveness, and poor judgement. Given these factors, the provision of appropriate diagnosis, interventions, and support services early in life and maintained throughout the life course is essential. Otherwise, many people with FASD are at high risk for becoming involved in the legal system, either as offenders or as victims. However, very little empirical evidence is available on the prevalence of FASD in correctional systems. There are no studies that have estimated the prevalence of FASD in correctional systems for any country other than Canada (5 studies: Burd, Selfridge, Klug, & Juelson, 2003; Fast, Conry, & Loock, 1999; Murphy & Chittenden, 2005; MacPherson, Chudley, & Grant, 2011; Rojas & Gretton, 2007) and the United States (1 study: Burd, Selfridge, Klug, & Bakko, 2004). In Canada, the prevalences of FAS and FASD have been reported to range from 0.1 per 1,000 (based on a survey conducted in federal and provincial prisons; Burd et al., 2003) to 10 per 1,000 (based on clinical assessment in an inpatient assessment unit of youth forensic psychiatric services; Fast et al., 1999); and from 98.9 per 1,000 (based on an interview and assessment in a male-only medium-security penitentiary for adults; MacPherson et al., 2011) to 233 per 1,000 (based on clinical assessment in an inpatient assessment unit of youth forensic psychiatric services; Fast et al., 1999), respectively. As reported in the only study conducted in the United States, which administered a survey in the prison systems and community corrections facilities, the prevalence of FAS was reported to be 0.0003 per 1,000 (Burd, Selfridge, et al., 2004). However, the authors of the study believed that this was an obvious underestimate.

Based on the available Canadian data, it has been estimated that youths with FASD are 19 times more likely to be incarcerated than youths without FASD in any given year (Popova, Lange, Mihic, Bekmuradov, & Rehm, 2011).

Morbidity (Primary Disabilities)

Prenatal alcohol exposure interferes with the normal developmental progression of the fetus, resulting in damage to the central nervous system, which is the underlying commonality of all diagnoses within the spectrum. Damage to the central nervous system is the most consistent expression of FASD across the spectrum of the disorder (Burd, Carlson, & Kerbeshian, 2009). The manifestations of brain damage are both age- and development-dependent. For example, a two-year-old with FASD is very unlikely to have a comorbid substance abuse disorder. However, the development of a substance abuse disorder is a huge issue for people affected by FASD in adolescence and adulthood.

The signs and symptoms commonly observed among people with FASD are often referred to as primary disabilities. The primary disabilities of FASD are those most closely related to the underlying central nervous system damage caused by prenatal exposure to alcohol, and include problems with adaptive behavior, attention, cognition, executive functioning, and memory. As a result, individuals with FASD may have trouble with abstract reasoning, organization, planning, understanding or recalling a sequence of events, and connecting cause-and-effect relationships. Another common characteristic of individuals diagnosed with FASD is dysmaturity. This term refers to widely varying levels of maturity in different areas of development, such as expressive language and language comprehension, social and self-care skills, and awareness and regulation of behaviors and emotions.

Notably, a recent study revealed that over 400 comorbid conditions co-occur among individuals with FASD. Some of these (e.g., language, auditory, visual, mental, and behavioral problems) are highly prevalent among individuals with FAS, ranging from 50–91% (Popova, Lange, Shield, et al., 2016). The most prevalent comorbid conditions found to occur among individuals with FAS are within the Congenital malformations, deformities and chromosomal abnormalities (Q00–Q99; 43%), and Mental and behavioural disorders (F00–F99; 18%) chapters of the International Statistical Classification of Diseases and Related Health Problems, 10th Revision (ICD-10; Figure 1). These impairments have lifelong implications.

Burden and Social Cost of Fetal Alcohol Spectrum Disorders

Figure 1 Percentage of comorbid conditions found to occur among individuals with FAS by ICD-10 chapter.

Abbreviations: Chapter II: Neoplasms (C00–D48); Chapter III: Diseases of the blood and blood-forming organs and certain disorders involving the immune mechanism (D50–D89); Chapter IV: Endocrine, nutritional and metabolic diseases (E00–E90); Chapter V: Mental and behavioural disorders (F00–F99); Chapter VI: Diseases of the nervous system (G00–G99); Chapter VII: Diseases of the eye and adnexa (H00–H59); Chapter VIII: Diseases of the ear and mastoid process (H60–H95); Chapter IX: Diseases of the circulatory system (I00–I99); Chapter X: Diseases of the respiratory system (J00–J99); Chapter XI: Diseases of the digestive system (K00–K93); Chapter XII: Diseases of the skin and subcutaneous tissue (L00–L99); Chapter XIII: Diseases of the musculoskeletal system and connective tissue (M00–M99); Chapter XIV: Diseases of the genitourinary system (N00–N99); Chapter XVI: Certain conditions originating in the perinatal period (P00–P96); Chapter XVII: Congenital malformations, deformations and chromosomal abnormalities (Q00–Q99); Chapter XVIII: Symptoms, signs and abnormal clinical and laboratory findings, not elsewhere classified (R00–R99); Chapter XX: External causes of morbidity and mortality (V01–Y98); Chapter XXI: Factors influencing health status and contact with health services (Z00–Z99); ICD-10: International Statistical Classification of Diseases and Related Health Problems, 10th Revision.

Mortality

As noted above, prenatal alcohol exposure is well known as a causal factor in pregnancy loss (Odendaal, Steyn, Elliot, & Burd, 2009). Current research demonstrates, not only increased rates of stillbirth and infant death (Odendaal et al., 2009), but also increased mortality risk across the lifespan (Burd et al., 2008). Specifically, Burd and associates (2008) found that individuals with FASD were at nearly five times the mortality risk than others of the same age and year of death, and that nearly half of all deaths occurred in young adulthood.

This increased mortality risk extends to siblings of a diagnosed child (whether or not they have a diagnosis of FASD themselves) and huge increases in mortality risk for the mothers of children with FASD have been reported. Burd, Klug, and Martsolf (2004) found a sibling mortality rate of 114 per 1,000 among children with FAS, compared to a sibling mortality rate of 19 per 1,000 among matched controls. Thus, the odds of sibling death among children with FAS were more than six times higher than the odds of sibling death in control children. Also, Li, Fisher, Peng, Williams, and Burd (2012), by using retrospective case-control methodology, estimated that mothers of children with FASD had a 44.8-fold increase in mortality risk compared to mothers of children without FASD. These findings suggest that a diagnosis of FASD is an important risk marker for premature mortality, not just for individuals with FASD, but for their siblings and mothers as well (Burd, Klug, et al., 2004; Li et al., 2012).

FASD is associated with a vast number and wide range of health and behavioral problems, and with increased premature mortality (shorter life expectancy and higher mortality rates), compared to the general population (Astley & Clarren, 1999; Burd, Klug, et al., 2004; Burd et al., 2008; Habbick, Nanson, Snyder, & Casey, 1997; Iyasu et al., 2002; Lemoine et al., 1968; Stratton et al., 1996). In a recent review of mortality among individuals with FASD, the five leading causes of death were malformations of the central nervous system, congenital cardiac abnormalities, sepsis, kidney malformations, and cancer (Thompson, Hackman, & Burd, 2014). This study also revealed that over half the reported deaths (54%) occurred in the first year of life. In addition, the attempted suicide rate is much higher among adults with FASD (23%; Streissguth, Barr, Kogan, & Bookstein, 1996) when compared to the general U.S. adult population’s rate (4.6%; Kessler, Borges, & Walters, 1999), and among persons with intellectual disabilities (11%; Hardan & Sahl, 1999; Lunsky, 2004). This could be because of difficulties with “fitting into” mainstream life; that is, the confluence of the neurodevelopmental deficits contributes to psychosocial and mental health problems at every developmental stage, with suicide attempts being among the most serious (Huggins, Grant, O’Malley, & Streissguth, 2008). Thus, FASD is a substantial burden to society in production terms due to the disabilities and premature mortality of members in the labor force affected by this disorder.

Secondary Disabilities

In addition to the primary disabilities faced by those with FASD, there are also secondary disabilities. Secondary disabilities (the disabilities that a person with FASD is not born with, but that develop as a result of the interaction between primary cognitive and neuropsychological disabilities and the social and physical environment) can become evident across the developmental trajectory of the affected person. The manifestation of these conditions affects nearly all areas of psychosocial development, and these disorders may be present for extended periods of time before they are linked to FASD. In many cases, the conditions appear early and simply change over the course of development, resulting in considerable personal, familial, and societal costs. Commonly observed impairments include mental illness, disrupted school experience (suspension, expulsion, and/or drop-out), poor academic achievement and school failure, involvement with the law (trouble with authorities, being charged with or convicted of a crime), confinement (inpatient treatment for mental health and/or substance use problems, or incarceration for crime), alcohol and other drug problems, sexually deviant behavior, problems with employment, and dependent living (Streissguth et al., 1996, 2004). These impairments appear to occur at a high rate among individuals with FASD. For example, it was estimated that approximately 37% of individuals with FASD either abuse or are addicted to alcohol and/or drugs (Popova, Lange, Burd, Urbanoski, & Rehm, 2013); that youths with FASD are 19 times more likely to be incarcerated than youths without FASD in a given year (Popova et al., 2011); and that approximately 59% of adults with FASD have had disrupted school experiences (Streissguth et al., 2004). When combined with primary disabilities, these secondary disabilities increase the complexity of care the person requires and result in significant social and economic costs to society.

However, certain protective factors such as early diagnosis and intervention, and providing an appropriate environment, improve outcomes and decrease the risk for secondary disabilities up to fourfold (Paintner, Williams, & Burd, 2012; Streissguth et al., 2004). Special education may be an important factor in reducing the occurrence of secondary disabilities (Rangmar et al., 2015).

Economic Cost of FASD

Challenges of Cost Studies on FASD

There are many challenges associated with estimating the burdens and costs associated with FASD, regardless of the country. First, the prevalence of FASD is currently unknown in most countries, either among the general population, special populations (e.g., children in care, criminal justice populations), or specific communities (e.g., Aboriginal populations). This significant gap in data highlights the need for accurate FASD-prevalence data, which would facilitate accurate epidemiological and cost estimates in the future. Second, whenever prevalence estimates are available in a population under study, economic estimates depend on the definitions and criteria used for diagnosing FASD, which are not always specified. It is unlikely that the estimates of the prevalence of FASD in the different service sectors are made using the same diagnostic criteria or guidelines, which could result in an error in the estimation of the number of individuals with FASD serviced within each sector, an error dependent on the specificity and sensitivity of the criteria/diagnostic guidelines used. Even though this is unavoidable, given the number of FASD diagnostic criteria/guidelines available, it must be recognized. Third, it is currently not possible to estimate several cost-drivers simply because the minimum required data are not available to make a comprehensive or reliable cost estimate. For instance, data on FASD within specific service sectors of interest (i.e., the number of individuals with FASD utilizing specific services) are completely absent. Therefore, there is an urgent need for a centralized reporting system in which FASD-specific data from various sectors can be stored and shared. Fourth, “FASD” captures only a portion of people affected by prenatal alcohol exposure. This is important, as a diagnosis of FASD sets a severity threshold that does not include a substantial number of people with adverse outcomes as a result of prenatal alcohol exposure. This has serious implications for cost studies, as the costs during pregnancy and prior to diagnosis cannot be captured. There are multiple constraints around using a diagnosis of FASD as the criterion for obtaining utilization data, including infrequent use of admission and discharge notes, as well as the difficulty of identifying diagnostic evaluations and linking them with other cost sources. Fifth, FASD is a familial disorder with high rates of recurrence. Referrals to diagnostic centers are typically made for individuals and often do not reflect the increase in risk for siblings, who are also likely to have FASD. Thus, in many cases, the referral for evaluation should include the entire sibship, including adults. The cost of several people having FASD in a single family clearly has important implications for productivity losses, for example.

In general, the lack of empirical methods for collecting cost and utilization data needs to be overcome before a full estimate can be made for any country. Since FASD has an impact on multiple systems, calculating the economic impact requires collaboration across many sectors and jurisdictions within a country. Thus, the scope and accuracy of the current cost estimates are contingent on data availability at the time of the estimation.

Existing Cost Studies with Comprehensive Methodology (by Country, in Alphabetical Order)

Canada (in Canadian Dollars)

The economic impact of FASD has been measured in eight Canada-based studies (Fuchs et al., 2009; Popova, Lange, Burd, & Rehm, 2016; Stade, Ungar, Stevens, Beyene, & Koren, 2006; Stade et al., 2009; Thanh, Jonsson, Dennett, & Jacobs, 2010; Thanh & Jonsson, 2009, 2014, 2015). Stade and colleagues (2006) measured the economic impact of FASD for 2003 using a modified version of the Health Services Utilization Inventory (Browne et al., 2001) on a sample of 148 parents (biological, adoptive, or foster) who were either living with or responsible for the care and welfare of a child with FASD between the ages of one and 21 with either FAS or FAE. At the patient level, the estimated total adjusted annual cost associated with FASD per child was $14,342 (95% CI: $12,986–$15,698). At the population level, using a conservative prevalence rate of 3 per 1,000, the cost of FASD annually was $344.2 million (95% CI: $311,664,000–$376,752,000). This prospective cross-sectional research study estimated costs from the perspectives of the society, the Ministry of Health, and the patient/family. The cost components included were: direct costs, such as medical care (hospital admissions, health professional services, and medication), educational services (home schooling, special schooling, and residential programs), social services (respite care, foster care, and legal aid), indirect costs (productivity losses; measured using the human capital approach), and other costs (out-of-pocket expenses).

Stade and colleagues (2009) later attempted to overcome the limitations of their previous study (Stade et al., 2006) by including the cost for infants from the day of birth to one year of age, the cost for adults beyond the age of 21 (up to the age of 53), and the cost of children residing in institutions (for a total of 250 participants with diagnosed FAS, pFAS, or ARND), as well as by adding additional cost components (residential programs, job education, institutionalization, and government pensions). The adjusted annual cost of FASD at the patient level, for 2007, was estimated as $21,642 (95% CI: $19,842–$24,041), while at the population level, the estimated adjusted annual cost was $5.3 billion (95% CI: $4.12 billion–$6.4 billion) for persons 0–53 years of age. In age-group–specific analyses, the authors found a decreasing cost per case with increasing age. Thanh et al. (2010) later recalculated the costs reported by Stade et al. (2009), adding costs for correctional services. Overall, the annual costs totaled $6.2 billion for all cases, with $700 million attributed to correctional services.

In an observational study, Fuchs et al. (2009) calculated incremental costs for children (defined as 0–21 years) with an FASD diagnosis who were permanent wards (i.e., children placed under protection of a legal guardian and the legal responsibility of the government) in Manitoba compared to the costs for a random sample of children from the general population. The Child and Family Services Administrative database was used to estimate the total number of cases, as well as the annual incremental costs per case in 2006. The authors found a total incremental annual cost of $6,416 per case, including $1,001 for healthcare (i.e., hospitalizations, ambulatory physician visits, and prescription medications), $5,166 for special education services, and $249 for subsidized child care services.

Thanh and Jonsson (2009) estimated the annual long- and short-term social cost of FASD in Alberta, Canada. The “annual long-term cost” refers to the projected amount of money incurred by the cohort of children born with FASD each year (lifetime cost), which was calculated based on a prevalence of 3 per 1,000 live births (lower rate) and 9 per 1,000 live births (upper rate) along with the total number of live births per year in 2002–2005. The “annual short-term cost” refers to the amount of money incurred by people presently living with FASD, calculated using the FASD cost calculator developed by the FAS Center at the University of Dakota. The cost calculator included the following cost components: healthcare costs, special education, juvenile justice services, adult corrections, and service delivery systems. The costs used in the calculator were 1996–1997 United States dollars, and were adjusted to 2008 Canadian dollars using a 5% discount rate and an exchange rate of 1.03. The lifetime cost of caring for each individual child born with FASD was estimated to be approximately $1.1 million (annual cost per child was taken from Stade et al., 2006; $15,812 multiplied by the average age at death—71.6). The total annual cost of FASD (short-term cost) for Alberta was estimated as ranging from $48 million (based on the lower prevalence rate) to $143 million (based on the upper prevalence rate), and the daily cost from $105,000 (based on the lower prevalence rate) to $316,000 (based on the upper prevalence rate). Notably, the long-term economic cost for the disorders associated with FASD rose from $130 to $400 million from 2002 to 2005, respectively.

One study investigated the cost of healthcare use associated with FAS and FASD (exclusive of FAS) for Alberta, Canada (Thanh & Jonsson, 2014). The total number of FAS cases and FASD-related conditions, as well as the associated costs, were derived from the Alberta Health databases of inpatients, outpatients, and physicians from the years 2003–2012. Alcohol-attributable fractions for FASD-related conditions such as learning disabilities, anomalies of the eyes, oppositional defiant disorder, or mental retardation were used to quantify costs for those conditions. Costs for medications were added as 30% of the total health service costs based on previous studies (Fuchs et al., 2009; Stade et al., 2009). All costs were calculated by sex and age group and converted to 2014 Canadian dollars. The total annual healthcare cost per person, including physician, outpatient and inpatient services, and medication was estimated to be $6,236 for FAS and $5,576 for FASD. The authors found higher healthcare costs for males (compared to females) and strong differences by age group. By adding costs across age groups, the authors found a lifetime cost per case of $506,000 for FAS and $245,000 for FASD. For all persons with FASD in Alberta (i.e., an estimated 46,000 persons), the healthcare costs were estimated to be $259 million per year.

A modeling study based on a systematic literature review and meta-analysis estimated the costs of FASD associated with the Canadian Criminal Justice System (Thanh & Jonsson, 2015). The costs of crime and expenditures of the Canadian Criminal Justice System were conceptualized as encompassing such expenses as policing, courts, corrections, and costs for victims of crime (including healthcare, loss of productivity, costs of stolen or damaged property, as well as third-party losses and costs). It was assumed that FASD equally contributed to each cost component. All costs were converted to 2014 Canadian dollars. The average prevalence of FASD in the criminal justice system was estimated as 101 per 1,000. The total cost for FASD associated with the criminal justice system in 2014 was estimated at $3.9 billion per year (ranging from $1.9 billion to $7.0 billion). This included $2,112 million for the criminal justice system, $1,591 million for victims, and $234 million for third-party costs. Adding this to the previous cost estimation of Stade et al. (2009) resulted in a total of $9.7 billion each year, with the largest share (40%) of the total cost attributed to the criminal justice system.

Recently, Popova, Lange, Burd, and colleagues (2016) conducted a cost-of-illness study to examine the impact of FASD on the material welfare of Canadian society by analyzing the direct costs of resources expended on healthcare, law enforcement, children and youth in care, special education, supportive housing, long-term care, prevention and research, as well as the indirect costs of productivity losses of individuals with FASD due to their increased morbidity and premature mortality. The authors employed an economic model developed to calculate a comprehensive, evidence-based picture of the economic impact of FASD in Canada (Popova, Stade, Lange, & Rehm, 2012). The estimation of each cost driver was based on its own methodology and set of assumptions, and expert opinions were sought wherever data was not readily available. Each cost component was estimated for the most recent year of available data (from 2008 to 2012). The costs were then adjusted for inflation to 2013, using the Bank of Canada inflation calculator. The cost associated with FASD totaled approximately $1.8 billion (from about $1.3 as the lower estimate, up to $2.3 billion as the upper estimate) in Canada in 2013. The highest contributor to the overall FASD-attributable cost was the cost of productivity losses due to morbidity and premature mortality, which accounted for 41% ($532 million to $1.2 billion) of the overall cost. The second highest contributor to the total cost was the cost of corrections, accounting for 29% ($378.3 million). The third highest contributor was the cost of healthcare at 10% ($128.5 to $226.3 million). Table 1 presents the cost estimates for Canada.

Table 1 Annual Cost (in Canadian Dollars) of FASD Reported in the Canadian Studies

Reference

Year of study; Province (if applicable)

Prevalence

Direct Costs (per person cost [unless indicated]; percentage of total cost)

Indirect costs (productivity losses)

Other costs

Total annual cost for all persons with FASD

Healthcare

Special education

Social services

Corrections

Total direct costs

Stade et al., 2006

2003

3/1,000 persons

$3,976; 30.3%

$4,275; 32.6%

$2,866; 21.9%

Not included

$11,117; 84.8%

$1,055; 8.1%

$936; 7.1% (out-of-pocket)

$344.2 M

Stade et al., 2009

2007

10/1,000 persons

$6,630; 35.0%

$5,260; 28.0%

$4,076; 18.8%

Not included

$15,965; 82%

$1,431; 6.6%

$2,814; 13% (out-of-pocket)

$5.3 B

Fuchs et al. (2009)a

2006; Manitoba

Not applicable

$1,001

$5,166

$249

Not included

$6,416

Not included

Not included

Not reported

Thanh & Jonsson, 2009b

2002–2005; Alberta (in 2008 CAD)

3–9/1,000 live births

30.3%

32.6%

21.9%

Not included

84.8%

8.1%

7.1% (out-of-pocket)

$126–$410 M (long-term cost);

$48–$143 M (short-term cost)

Thanh & Jonsson (2014)

2003–2012; Alberta (in 2014 CAD)

46,000 people living with FASD

FAS: $6,236;

FASD: $5,576

Not included

Not included

Not included

Not included

Not included

$259 M

Thanh & Jonsson (2015)c

2014

101/1,000 persons in the criminal justice system

$2 Bd; 21%

$1.6 Bd; 17%

$1.3 Bd; 13%

$3.9 Bd; 40%

$8.8 Bd; 91%

Not included

$ 0.9 Bd; 9% (out-of-pocket)

$9.7 B

Popova, Lange, Burd et al., 2016e

2013

Specific to each cost component

$128.5 Md; 10.2%

$54.2 Md; 4.3%

$160.3 Md; 12.7% (children and youth in care, supportive housing, long-term care)

$378.3 Md; 30.0%

$728.8 Md; 57.8%

$531.9 Md; 42.2%

$7.5 Md; 0.6% (direct cost: prevention & research)

$1.3–$2.3 B

(a) Included only children who were permanent wards, and refers to incremental costs compared to the general population.

(b) Cost data and respective percentages were obtained from Stade et al. (2006).

(c) All estimates except for those for the criminal justice system were based on Stade et al. (2009).

(d) Cost for all persons with FASD.

(e) Costs based on lower estimates.

Abbreviations: B: billion; CAD: Canadian dollars; FAS: Fetal Alcohol Syndrome; FASD: Fetal Alcohol Spectrum Disorder; M: million.

South Africa (in United States Dollars)

A cross-sectional analytical study conducted by Credé, Sinanovic, Adnams, and London (2011) using an interviewer-administered questionnaire amongst caregivers of children (0–12 years of age) with FAS/pFAS in the Western Cape, South Africa, estimated the utilization of healthcare services, the annual direct and indirect healthcare costs per child, and the total cost to society for providing healthcare services to children with FAS/pFAS. Accordingly, it was found that the median number of annual visits to public healthcare facilities per child was eight. The total average annual cost per child was estimated to be $1,039 (95% CI: $809–$1,270), and the total annual societal cost was estimated to be $70,960,055 (95% CI: $5,528,895–$86,709,971).

Sweden (in Swedish Kronor)

As part of a larger study on the societal cost of alcohol consumption, Johansson and colleagues (2006) estimated the total annual cost of inpatient and outpatient care due to FAS to be $185,716 (inpatient care costs: $101,286, and outpatient care costs: $84,430) in 2002.

United States (in U.S. Dollars)

The economic impact of FAS has been estimated in ten America-based studies (Lupton, Burd, & Harwood, 2004; Abel & Sokol, 1987, 1991a,b; Harwood, 2000, 2003; Harwood, Napolitano, & Kristiansen, 1984; Harwood, Fountain, & Livermore, 1998; Rice, Kelman, & Miller, 1990; Rice, 1993; Weeks, 1989), two of which have estimated the total lifetime cost for a person with FAS (Harwood et al., 1984; Weeks, 1989).

Using a societal perspective and previously published data (Russell, 1980), Harwood et al. (1984; dual publication: Harwood & Napolitano, 1985) estimated the cost of FAS in 1980 using alternative FAS prevalence rates (based on a review of prospective studies) of 1.0, 5.0, and 1.67 per 1,000 live births, respectively, and included estimates of medical treatment, home and residential care, special educational services, and productivity losses for individuals with FAS from 0–65 years of age. The cost of FAS in 1980 ranged from $1.937 to $9.687 billion, with a median estimate of $3.236 billion. The total lifetime cost was estimated at $596,000 per individual. Based on this lifetime estimate, Lupton et al. (2004) demonstrated how the cost for one case of FAS is spread out over the person’s lifetime. After adjusting for inflation, the lifetime cost ($596,000) in 1980 became $2 million in 2002 ($1.6 million [80%] for healthcare, special education, and residential care for individuals with intellectual disability; and $0.4 million [20%] for productivity losses; Lupton et al., 2004). Furthermore, the cumulative cost of one case of FAS to age 65 would be $130,000 in the first five years, $360,000 in 10 years, $587,000 in 15 years, and more than $1 million in 30 years (Lupton et al., 2004).

Abel and Sokol (1987) measured the economic cost of FAS from the perspective of the healthcare system from birth to 21 years of age, and estimated that the economic burden of FAS in the United States was $321 million in 1984, using a prevalence of 1.9 per 1,000 (obtained by taking the average of several prospective and retrospective studies). The study estimated the cost of providing specialized services for pre- and post-natal growth retardation requiring neonatal intensive care, surgical repair of FAS-related birth defects and subsequent treatment, care for FAS patients with moderate or severe cognitive disabilities, and the cost of semi-independent supervised support for mildly cognitively disabled patients with FAS.

In 1991, Abel and Sokol (1991a) revised their previous estimate, using a more conservative prevalence of 0.33 per 1,000 (obtained from prospective studies that were comprised of primarily Caucasian samples and did not include any other racial/ethnic groups—e.g., Native Americans). Accordingly, this study produced a much lower annual cost estimate—$74.6 million. The majority of this economic burden (greater than 77%) was associated with residential care due to intellectual disability. However, Abel and Sokol (1991b) further refined their methodology (and the cost components that were included/excluded), and estimated an annual cost of $250 million (again, based on a prevalence of 1.9 per 1,000).

Again, from the perspective of the healthcare system, Rice et al. (1990; dual publication Rice, Kelman, & Miller, 1991), using the method of Abel and Sokol (1987), estimated the annual cost of treating birth defects associated with FAS in the United States in 1985 at $1.6 billion, based on a prevalence of 1.9 FAS cases per 1,000 live births. The cost drivers included the cost of care for FAS-related birth defects and cognitive disability, and residential care for individuals with intellectual disability between 21–65 years of age. Subsequently, based on increasing population size and healthcare costs from 1985 to 1990, Rice (1993) estimated an annual cost of $2.1 billion in 1990.

Harwood et al. (1998) estimated the annual cost to be $1.9 billion in 1992, based on a prevalence of 2.0 per 1,000. This estimate included the cost of treatment and care services to age 21, home and residential care to age 65 of people with moderate to severe intellectual disability, special education services, and productivity losses. Next, Harwood (2000, 2003) updated the 1992 cost estimate by adjusting for the change in national healthcare expenditures, the consumer price index for medical services, the adult population, and the hourly compensation index for productivity losses, which resulted in an estimate of $4.2 billion in 1998 (direct cost—$2.9 billion, indirect cost—$1.3 billion), and $5.4 billion in 2003 (direct cost—$3.9 billion [annual increase of 6.1%]; indirect cost—$1.5 billion [an annual increase of 4%]).

A study by Weeks (1989) reported that the lifetime cost for each child born with FAS was $1.4 million in Alaska in 1988. This study adapted the methodology used by Harwood and Napolitano (1985) and included the following cost components: developmental disability services, special education, social service costs, adult vocational services, and institutional care for intellectual disabilities to age 65.

More recently, Amendah, Grosse, and Bertrand (2011) calculated the medical expenditures for pediatric Medicaid enrollees with FAS in the United States for 2003–2005. Children with FAS incurred annual mean medical expenditures that were nine times higher than those of children without FAS during 2005 ($16,782 vs. $1,859). Table 2 presents the cost estimates for the United States.

Table 2 Annual Cost (in United States Dollars) of FASD Reported in the American Studies

Reference

Year of study; State (if applicable)

Prevalence (per 1,000)

Direct Costs (cost; percentage of total cost)

Indirect costs (productivity losses)

Other costs

Total annual cost for all persons with FASD

Healthcare

Special education

Residential care & home care

Total direct costs

Harwood et al., 1984; Harwood & Napolitano, 1985

1980

1.0; 5.0; 1.67a

$699 M (children: $125 M; adults: $574 M); 22%

$990 M; 31%

HC & RC with day services: $694 M; 22%

$2.4 B; 75%

$853.3 M; 25%

Not included

$3.2 B

Abel & Sokol, 1987

1984

1.9

Growth retardation: $118 M; Cleft palate, Tetralogy of Fallot & sensorineural anomalies: $18 M; 42%

Not included

24-hr RC due to ID: $109 M; semi-independent supervised support: $75.8 M; 58%

$321 M

Not included

Not included

$321 M

Weeks, 1989

1988; Alaska

1.67

Included

Included

Included

n/a

Included

Not included

$1.4 M (lifetime cost per individual)

Abel & Sokol, 1991a (revision of 1987 study)

1984

0.33

Treatment cost: $16.9 M; 22.7%

Not included

24-hr RC due to ID: $57.7 M; 77.3%

$74.6 M

Not included

Not included

$74.6 M

Abel & Sokol, 1991b (revision of 1987 study)

1987

1.9

Treatment cost: $104.5 M; 42%

Not included

24-hr RC due to ID: $145.2 M; 58%

$249.7 M

Not included

Not included

$249.7 M

Rice et al., 1990, 1991

1985

1.9

Treatment: $135 M; 8.4%

Not included

RC for 21+ yrs $1,287 B; 79.9%;

Full time RC <21 yrs $110 M, 6.8%; semi-independent supervised care $76 M, 4.7%

$1.6 B

Not included

$3 M; 0.2% (research)

$1.6 B

Rice, 1993 (update of Rice et al., 1990; 1991)

1990

1.9

Included

Not included

Included

n/a

Not included

Included

$2.1 B

Harwood et al., 1998 (update of Harwood et al., 1984, and Harwood & Napolitano, 1985)

1992

2

Included

Included

Included

n/a

Included

Not included

$1.9 B

Harwood, 2000 (update of Harwood et al., 1998)

1998

2

Included

Included

Included

$2.9 B

$1.25 B

Not included

$4.2 B

Harwood, 2003 (update of Harwood, 2000)

2003

2

Included

Included

Included

$3.9 B

$1.5 B

Not included

$5.4 B

(a) Estimates are based on a prevalence of 1.67 per 1,000.

Abbreviations: B: billion; FASD: Fetal Alcohol Spectrum Disorder; HC: home care; I: incidence; ID: intellectual disability; M: million; n/a: not available; RC: residential care.

Prevention of FASD

FASD affects from 1–5% of live births in the developed world (May et al., 2015). Therefore, every year, hundreds of thousands of children are born with a lifelong condition with a known and preventable cause. In many countries, less than one percent of the cost of caring for people with FASD is invested in prevention. One way to conceptualize the value of prevention would be to consider the cost of FASD. If prepayment of the cost of care were required, developed nations would need to deposit $540,000 on the day before the birth of every child with FASD (Lupton et al., 2004).

Prevention strategies should emphasize the detection of alcohol abuse prior to pregnancy as the primary level of prevention. Effective treatment of any identified cases of alcohol dependence/use disorders could reduce the risk of FASD. A second level of prevention would focus on the detection of prenatal alcohol use. Given that brain damage appears to be the result of repeated exposure of the developing fetal brain to alcohol, it is likely that some degree of risk reduction would accrue from reducing the number of exposure episodes, the magnitude of exposure (drinks per drinking day), and cumulative exposure during pregnancy. One model of prevention, as currently used in North Dakota, is to have every prenatal care site utilize a standardized screen for prenatal alcohol exposure (see Figure 2). The One-Question-Screen reduces provider burden by having them ask only one question (with follow-up questions required only if exposure is reported), rather than several (it should be noted that, in settings other than a prenatal care setting, a second question may be required—e.g., When did you become pregnant?). Preliminary data suggest that this strategy detects as many (if not more) women who drink as other commonly used prenatal screens (Williams, Nkombo, Nkodia, Nkodia, & Burd, 2013). This strategy also emphasizes the assessment and recording of exposure dosimetry for women who are drinking. It should be noted that there are several other brief assessments that have been shown to have good sensitivity and are widely used to screen for prenatal alcohol use (e.g., the Alcohol Use Disorders Identification Test–Consumption [AUDIT-C; Bush, Kivlahan, McDonell, Fihn, & Bradley, 1998], CRAFFT screening interview [Knight, Sherritt, Shrier, Harris, & Chang, 2002], 4P’s Plus screen [Chasnoff et al., 2005], and the Substance Use Risk Profile-Pregnancy scale [Yonkers et al., 2010]).

Burden and Social Cost of Fetal Alcohol Spectrum Disorders

Figure 2 The One-Question-Screen

Abbreviation: PAE: Prenatal alcohol exposure

The development of prevention strategies should include a discussion of four strategies. These strategies all build on existing services that are typically available in prenatal care settings, delivery hospitals, child care settings, and substance abuse treatment settings.

Firstly, recurrence prevention is included because the risk for FASD is increased for younger siblings within a sibship. Thus, if a woman can reduce her alcohol use or quit drinking, any subsequent pregnancies will have decreased exposure, and the risk of FASD will be reduced or eliminated entirely. Secondly, substance abuse treatment programs for young women, often with young children, can be difficult to access. Improving access to treatment services, improving the effectiveness of substance abuse treatment, and offering comprehensive case-management and recovery support services for pregnant and postpartum women can have important community, as well as individual, benefits (Substance Abuse and Mental Health Services Administration [SAMHSA], 2009). For example, the Parent-Child Assistance Program (PCAP) is an intensive case-management model developed with the primary aim of preventing future alcohol- and drug-exposed births (and thus, FASD) among high-risk women (Ernst, Grant, Streissguth, & Sampson, 1999; Grant, Ernst, Pagalilauan, & Streissguth, 2003; Grant, Ernst, Streissguth, & Stark, 2005; Grant et al., 2011; Grant, Graham, Ernst, Peavy, & Brown, 2014); the model has been replicated throughout Washington State and at 40 sites in Canada. Economists recently estimated the cost effectiveness of PCAP in Alberta, Canada, and reported that the program prevented approximately 31 cases of FASD among 366 clients in a three-year period, with a significant net monetary benefit of approximately $22 million (range $13–$31 million; Thanh et al., 2015). Thirdly, many women who will have several children will quit drinking as soon as they find out they are pregnant. They should be encouraged to not starting drinking again, especially through their childbearing years. Fourthly, as discussed above, implementing prenatal screening programs can be simple and inexpensive.

Conclusions

Challenges

Diagnosis

The identification of a child exposed to alcohol in utero is crucial, as it can lead to close monitoring of his/her development, can facilitate early diagnosis, and can, if necessary, lead to the implementation of timely interventions, which are key to improving the quality of life of individuals with FASD, potentially preventing secondary disabilities. Furthermore, early diagnosis could prevent subsequent alcohol-exposed births by providing appropriate interventions, treatment, counseling, and support to the birth mothers (Astley, Bailey, Talbot, & Clarren, 2002). However, given that individuals with FASD can present with myriad deficits and disabilities, the diagnosis of FASD is challenging. Determining whether diffuse brain damage is the explanation for the observable behavioral problems of a person with FASD, and confirming that this damage is more likely to be due to prenatal alcohol exposure than to other causes, is not a simple task.

The challenges of diagnosing FASD are exacerbated by a lack of uniformly accepted diagnostic criteria for FASD. While the current criteria considerably overlap with one another, there is limited guidance for clinicians in selecting the optimal criteria, as there is a general lack of studies comparing the specificity, sensitivity, and accuracy of the various criteria (Burd, Klug, Li, Kerbeshian, & Martsolf, 2010). Ultimately, the absence of a standardized set of criteria can lead to diagnostic misclassification (Astley & Clarren, 2000), which itself has a number of consequences. Specifically, it can result in inaccurate prevalence estimates, inappropriate treatments and interventions for patients, and from a clinical research point of view, the inability to detect a clinically meaningful difference between two groups (Astley & Clarren, 2000). Alarmingly, it has been argued that nearly all FASD is undiagnosed, even in clinical settings where FASD is an important area of emphasis (Chasnoff, Wells, & King, 2015).

Ascertainment of Prevalence

In addition to the challenges faced in regard to diagnosis, the different existing methods of surveillance can produce apparent discrepancies in the prevalence estimates obtained, with each approach yielding different data sets with their own implications and limitations (May & Gossage, 2001). The three main approaches to studying the prevalence of FASD are: (1) passive surveillance, (2) clinic-based studies, and (3) active-case ascertainment (ACA). Passive surveillance is the use of existing record collections (e.g., birth certificates, special registries, medical charts, adoption records) in a particular geographical catchment area to look for documented or probable cases of FASD. Clinic-based studies are prospectively conducted in prenatal clinics or hospitals. At-risk newborns are identified by screening biological mothers, and are examined and diagnosed if they are shown to have FASD. ACA is an approach whereby researchers actively seek, find, and recruit children who may have FASD within a given population. Once these children are identified as possible cases, clinical specialists examine these cases and assess the children for a final diagnosis of FASD. There are also mixed-method approaches, which utilize two of these three approaches (usually ACA and passive surveillance). The different epidemiological surveillance methods described above tend to provide different prevalence estimates, with ACA producing the highest prevalence estimates (May & Gossage, 2001; May et al., 2009).

Putting the Cost of FASD into Perspective

The authors of this paper have estimated the cost of FASD in Canada to be approximately $1.8 billion Canadian dollars per year ($1.3 as the lower estimate, to $2.3 billion as the upper estimate; Popova, Lange, Burd, et al., 2016). This translates to:

  • $205,362 per hour

  • $4,931,000 per day

  • $34,615,000 per week

  • $150,000,000 per month

  • $1,800,000,000 per year

  • $9.0 billion over the next five years

Future Directions

Increased attention needs to be given to tracking people with FASD across systems of care so we can improve our knowledge of the costs incurred by and service needs for this population. Increased emphasis needs to be given to FASD-related mortality for mothers, children diagnosed with FASD, and their siblings. This appears to be especially important in the developing world. When the cost of care for a potentially preventable disorder runs in the millions, it should be seen as an indication that it is time to start increasing funding toward the development of new prevention strategies. This should include a dramatic increase in emphasis on screening children whose mothers are in substance abuse treatment, and every child entering foster care or the juvenile correctional systems.

Currently, there is no known strategy to diagnose thousands of people in the geriatric population with FASD, and little to no knowledge of the developmental implications of FASD in the elderly. As we follow people with FASD into their 60s and 70s, and even a few into their 80s, premature mortality and early-onset dementia are two common problems that are encountered.

Improved treatment programs for adults with FASD are urgently needed (Moore & Riley, 2015; Reid et al., 2015). Can we develop a cost-effective model for the long-term management of these people? Some evaluation endpoints are straightforward, and include comparisons of rates of substance abuse, hospitalization rates for mental disorders, and entry into correctional systems. When we consider these endpoints, it seems possible that we could first consider the adoption of some existing models of care, with a few modifications. We also need models for the improved treatment of young children with FASD in preschool, elementary school, and middle and high school settings. These models need to have large treatment effects if they are to be useful. Lastly, although it may sound simplistic, we need to expand existing models such as PCAP and others (Child Welfare Information Gateway, 2014; DiLorenzo, 2013), which identify mothers of children with FASD and get them into effective treatment programs.

In conclusion, FASD is a paradox. The impact and cost of FASD is complex and wide-ranging, yet studies indicate that, if a program is able to prevent just one case of FASD, the results can be considerable. FASD is a largely preventable range of disorders, for which thinking small can produce big results.

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