Societal costs of fetal alcohol syndrome in Sweden

Fetal alcohol syndrome (FAS) was first described in 1973 [1, 2] and is caused by maternal alcohol consumption during pregnancy. Alcohol is a teratogen that easily crosses the placenta and thus can have devastating effects on the developing embryo and fetus [3, 4]. The brain is particularly vulnerable to prenatal alcohol exposure [5] and the most profound effects are on brain development and on cognitive and behavioural outcomes [3, 4]. There is no safe “lower limit” for alcohol consumption during pregnancy and the absolute amount of alcohol that will not cause damage to the developing brain is not known. Prenatal exposure to alcohol can disrupt fetal brain development at any point during pregnancy [6] and the disturbances are more frequent and severe if the mother abuses alcohol throughout pregnancy [7, 8]. A recent Swedish study reported that relatively few women continue to drink alcohol during pregnancy. Nevertheless, 84 % of the women reported drinking in the year preceding pregnancy and most of these women continued to drink until pregnancy recognition [9].

The diagnosis of FAS has been expanded and refined and there are currently four widely used criteria. These diagnostic criteria have some differences, yet rely on anomalies in three distinct areas; pre- and/or postnatal growth deficiency, central nervous system dysfunction, and characteristic facial anomalies [10, 11, 12, 13] (for review see [4]). Whether there also needs to be a confirmed maternal alcohol exposure varies between different diagnostic criteria [4]. Prenatal alcohol exposure causes a wide range of neuropsychological and behavioural disabilities and to identify all these different outcomes the non-diagnostic umbrella term “fetal alcohol spectrum disorders” (FASD), where the full-blown FAS is included, has come into use [4]. FAS is the only diagnosis defined according to the International Classification of Diseases (ICD-10; code Q86.0) [14].

FAS is recognized as the most common identifiable cause of acquired mental intellectual development disorder [3]. The true prevalence of FAS is not known. The latest prevalence study in Sweden was performed in the late 1970s and reported an incidence rate of fetal alcohol lesion of one per 300 deliveries of whom half had the complete FAS [15], corresponding to a prevalence of FAS of 0.2–0.3 % (or 19,000–29,000 individuals). Recent in-school studies estimated the prevalence of FAS at 0.2–0.9 % in the United States [16, 17] and 5.9–9.1 % in a South African community [18].

Brain dysfunction in children with FAS often leads to primary disabilities such as poor adaptive functioning, language deficits, attention difficulties and reasoning and memory deficits [6]. The neuropsychological and neurobehavioural problems often stem from early childhood, during adolescence and into adulthood [5]. Secondary disabilities that arise after birth as a result of the primary disabilities include psychiatric disorders, disrupted school and employment experiences, alcohol abuse/illicit drug use, trouble with the law and inappropriate sexual behaviour [6, 19]. A Swedish research group found that adults who were diagnosed with FAS in childhood revealed impaired psychosocial outcomes [20], decreased cognitive and executive functions and impaired social cognition [21]. Furthermore, individuals with FAS often have problems with their daily planning and living an independent life [3, 6].

However, adverse life outcomes in terms of secondary disabilities may be limited by protective factors such as receiving the diagnosis of FAS at an early age, placement in a stable environment and appropriate interventions for primary and secondary disabilities [6, 19]. In addition, children with FASD who were placed at an early age in a family home (formerly foster home), were seen to have a decreased risk of developing secondary disabilities [22]. Placement in a family home has also been suggested to lead to improved overall performance and a better quality of life for affected children in Sweden, although normalization does not occur [7, 8]. Special education and assistance in Swedish schools are also factors that are believed to limit the secondary disabilities [20].

Estimates of the economic impact of FAS/FASD demonstrate a significant cost burden on the individual, family and society. Unfortunately, health economic studies of FAS/FASD are sparse and the majority have been conducted in Canada and the United States [23]. Although these studies included many cost components, the total cost of prenatal alcohol exposure is underestimated [24]. Currently, to the best of our knowledge, there is a lack of cost calculations of FAS/FASD from a Swedish perspective.

The aim of this study was to perform a health economic calculation of the annual societal cost of FAS in Sweden, focusing on the secondary disabilities thought feasible to limit via early interventions. The present study is part of a major initiative in Sweden in the area of children as next of kin and was commissioned by the Swedish National Board of Health and Welfare. The study focuses on the economic cost of FAS in Sweden, in the light of relevant international studies.

This study is a prevalence-based cost-of-illness analysis. Both the bottom-down approach, where costs for a defined subpopulation were estimated and then extrapolated to represent the entire population, and the top-down approach, where national frequencies and events formed the basis for estimates, was used [25, 26].

The present calculations are based on Swedish studies and Swedish cost data. Information that was otherwise inaccessible and/or unreliable was obtained from relevant experts such as a paediatrician, child psychiatrist, child welfare officer, care administrator, family members/relatives of individuals with FAS, researchers and staff at special accommodation facilities.

The cost of FAS was calculated as the annual total societal cost and the annual additional costs of FAS, based on the difference between the cost in the FAS group and a comparison group. The calculations are based on individuals up to the age of 64 years, and are also divided by age into children (0–17 years) and adults (18–64 years).

All costs were estimated in Swedish Crowns (SEK) according to 2014 prices and then converted to euros (€1.00 = SEK9.097) and, where applicable, the consumer price index was applied as the conversion factor.

To the best of our knowledge this is the first health economic study of the societal cost of FAS in Sweden. The calculations are based on the secondary disabilities that develop in varying degrees in individuals with FAS. The occurrence of secondary disabilities was adapted from a Swedish register-based long-term follow-up study of psychosocial outcomes in adults with a verified diagnosis of FAS [20]. The annual total societal cost of FAS was estimated at about €76,000 per child and €110,000 per adult, and the major proportion was represented by direct costs (82 and 74 %, respectively). Applying a prevalence of FAS of 0.2 % (corresponding to ~19,000 individuals in the entire population), the total societal cost was estimated at €1.6 billion per year in the Swedish population. This assumption is based on a previous Swedish study [15] and is deemed low compared with international studies from e.g. the United States (0.2–0.9 %), but within the interval [16, 17]. To the best of our knowledge, there are no additional published prevalence studies in Sweden. The major cost driver was the cost of societal support, both in children and adults with FAS. The direct costs represented the highest proportion of the total cost both in children and in adults with FAS, whereas there were only minor differences in the proportion of direct and indirect costs in the comparison group.

A complicating factor in this study was the actual number of children and adults with alcohol damage to date in the society. Often adverse life outcomes are not restricted to those with or without the classical facial features of FAS or to those with or without mental retardation [19]. Not all children born with alcohol damage have the typical external features (facial anomalies) of FAS, but instead they have disabilities that are often difficult to diagnose, leading to an estimated number of unknown cases [13].

The secondary disabilities often lead to major societal costs and may be limited by early interventions (from the society). Protective factors helping children with FAS avoid adverse life outcomes include receiving the diagnosis of FAS at an early age, placement in a stable environment and appropriate interventions for primary and secondary disabilities [6, 19]. International cost studies have shown that prenatal alcohol exposure is associated with a major health care and cost burden on the society [37, 38, 39, 40]. For example, the total direct cost of FAS in Canada in 2008–2009, was estimated at about €5.3 million (authors’ recalculation from Canadian dollars to euros at 2014 cost level) [37]. In another Canadian study, the total adjusted annual costs associated with FASD were estimated at ~€16,000 per individual [39]. Furthermore, the annual cost of care among children with FASD ranged from €43.3 to €148.7 million [38]. It is, however, difficult to compare the results from different cost studies, since the methodology and population often varies between the studies.

In this study, societal support represented the highest portion of the total cost based on ~80 % of children growing up in some form of special accommodation, but with unknown distribution between family home and residential care. It was, however, assumed that the majority of children with FAS grow up in a family home, which was also reported in a Swedish follow-up study of children of alcoholic mothers [7, 8] where early placement in a family home was suggested to lead to improved performance and enhanced quality of life [7]. Similarly, early placement of children with FASD in a family home was able to limit disabilities, such as socio-emotional problems (including neuropsychological problems) [22]. In contrast, a further Swedish long-term follow-up study, reported that children placed in out-of-home care showed a higher risk of psychiatric problems, criminality and low educational levels, and that these problems were higher in children placed for their behavioural problems [41]. The prognosis of individuals with FAS is in some cases relatively optimistic, since a child with FAS in Sweden at risk of growing up in an inappropriate environment, with e.g. abuse, is routinely taken care of and generally receives correct care early [20], compared with children taken care of during adolescence due to their own behavioural problems [41]. Placement in a family home can thus be seen as an unavoidable cost, yet an important intervention commonly preventing serious problems.

Due to their limited basic intelligence, a high proportion of children with FAS/FASD attend school for children with special educational needs [7, 42]. Since many of these children also have behavioural difficulties, even those children with normal intelligence often end up in special school [42]. However, some children attend normal school with extra support, such as a special needs teacher and/or personal assistance. In the study by Rangmar and colleagues, a quarter of children with FAS had attended a special needs school (primary school level) and only about 5 % had secured a postsecondary qualification [20]. In Sweden, the cost of special school is approximately five times higher than normal school [29]. Additional costs associated with normal school attendance includes, for example, extra support, but the extent of such support within the FAS population is not known. However, the individuals with FAS who had attended normal school most likely had had access to personal assistance at school, at least for a limited period. It can be argued that, in the long term, it should not solely be a discussion about the costs, since the opportunity of attending special school may result in individuals with FAS being able to work, which is positive both for the individual (quality of life) concerned and from a societal perspective. The cost of special school is thus difficult to avoid since adapted schooling is a necessary early intervention that may prevent other costs later in life.

Mental illness is relatively common among individuals with FAS/FASD [20, 21, 43] and the problems often persist throughout life [20, 42]. The cost of psychiatric disorders among adults with FAS was only a minor part of the total societal cost. This can partly be explained by the exclusion of productivity loss from the cost of psychiatric disorders, since productivity loss was already included in the calculation of reduced working capacity. In addition, the calculation of the cost of psychiatric disorders was based on four diagnoses (depression, bipolar disorder, generalized anxiety disorder and schizophrenia) [30], which were deemed to be representative of reflecting mental illness. The cost of psychiatric disorders was only estimated for adults, but the majority of these individuals were probably also treated during childhood and/or adolescence. Nevertheless, information about the need for psychiatric care among children/adolescents was lacking.

Individuals with FAS are at increased risk of developing problems with alcohol and/or illicit drugs [19]. However, this was not so apparent in the long-term study by Rangmar et al. [20]. The societal cost of alcohol/drug abuse was thus not so high, although not negligible, as it constituted 15 % of the annual total societal cost among adults with FAS. Alcohol abuse and/or illicit drug use often leads to criminality and individuals with FAS may easily find themselves falling into trouble with the law [6]. Canadian data highlighted that youths with FASD are 19 times more likely to be in prison than youths without FASD [44]. Comparable Swedish data is not available. Besides, the cost of criminality was already included in the cost reference for alcohol/drug abuse [34].

The working capacity of individuals with FAS is often reduced, resulting in unemployment, disability pension and/or social welfare payments. Half of the individuals with FAS in the Swedish study were unemployed [20] resulting in a productivity loss corresponding to a quarter of the total societal cost. At the same time, half of the individuals with FAS were employed; nevertheless, the disposable income indicated lower-paid jobs [20]. Even though these jobs were supplemented by welfare benefits, it can be argued that it is positive from a societal perspective to be self-sufficient.

The caring support to individuals with FAS from family members/relatives is of high intensity, due to the disabilities that commonly develop in FAS as a result of brain damage caused by prenatal alcohol exposure. The productivity loss among parents/foster parents, who often have to take on board considerable responsibility as informal carers, is calculated as almost one fifth (18 %) of the societal cost among children with FAS (based on informal caring by one parent per family). This estimation was based on 25 % reduced working time (in both groups) when the child is below 18 years of age, with the distinction that all individuals in the FAS group need informal caring compared with around one fifth in the general population. This can be compared with a Swedish study about the burden of informal caring of patients with psychoses, showing that informal carers spent 22.5 h per week on caring activities and about 14 % of their gross income on care-related activities [45].