Full length articleThe continuum of fetal alcohol spectrum disorders in four rural communities in south africa: Prevalence and characteristics
Introduction
The diagnosis of fetal alcohol spectrum disorders (FASD) has been evolving since the first criteria for fetal alcohol syndrome (FAS) were defined by Jones and Smith (1973). Children with the most dysmorphic features and cognitive and behavioral impairments were identified as FAS. Soon thereafter, less consistent, less severe patterns of dysmorphia and impairment were recognized in animal and human studies and referred to as fetal alcohol effects (FAE; Aase, 1994, Aase et al., 1995, Clarren and Smith, 1978). In 1996, four specific diagnoses were developed by a committee of the Institute of Medicine (IOM): FAS, partial FAS (PFAS), alcohol-related birth defects (ARBD), and alcohol-related neurodevelopmental disorder (ARND) (Stratton et al., 1996). These four diagnoses form a continuum from the most dysmorphic to least dysmorphic and came to be known as FASD (Streissguth and O’Malley, 2000, Warren et al., 2004). FASD are rarely diagnosed or diagnosed properly (Chasnoff et al., 2015) which colors understanding of the full spectrum of FASD. Because of this, FASD epidemiology information gathered through registries are inaccurate (Fox et al., 2015). Focused epidemiologic studies of FASD are difficult and expensive to pursue (Stratton et al., 1996). Few studies have documented the prevalence and characteristics of the full continuum of FASD, especially in the United States and Europe where FASD were believed for many years to be rare occurrences, affecting an estimated maximum of 1% of the general population (Sampson et al., 1997). But recent studies indicate that FASD prevalence is much higher, 2–5%. At least this has found to be true in the USA, Italy, Poland, and Croatia (May et al., 2009, May et al., 2011a, May et al., 2014a, May et al., 2015, Okulicz-Kozaryn et al., 2015, Petković and Barišić, 2010, Petković and Barišić, 2013), and especially true in South Africa.
Unfortunately, some communities in South Africa (ZA) have the highest general-population rates of FASD anywhere in the world. But from an epidemiologic perspective, ZA has proven to be an excellent venue for understanding the diagnosis, prevalence, characteristics, and etiology of the FASD continuum. The prevalence and characteristics of FASD have been described by the authors of this paper in four studies of a single municipality and surrounding rural areas in the western portion of the Western Cape Province (WCP) close to the Cape Town metropolitan area (May et al., 2000, May et al., 2007, May et al., 2013a, Viljoen et al., 2005). Other researchers have completed community studies in other provinces of ZA (Oliver et al., 2013, Urban et al., 2008, Urban et al., 2015, Viljoen and Hymbaugh, 2003). In the previous case control studies in the WCP, physical and neurobehavioral traits were researched extensively, the diagnostic categories have been explored with thousands of children, and research has progressed from an exclusive focus on FAS to delineating and understanding all forms of FASD. In a recent WCP study publication, FAS affected 59–91 per 1000 children, PFAS 45–70 per 1000, no cases of ARBD were found, and ARND affected 31–47 per 1000. The total FASD rates were 135–208 per 1000 or 13.5–20.7% (May et al., 2013a). Quite noteworthy, the prevalence of FASD was highest in rural areas surrounding the town in all previous studies in the WCP. Multiple other findings indicated that norms and practices of regular binge drinking, low socioeconomic status (SES), insufficient nutrition, high fertility, and challenging conditions for prenatal and postnatal development combine to elevate the prevalence and severity of FASD (May et al., 2005, May et al., 2008, May et al., 2013b, May et al., 2014b, Viljoen et al., 2002).
As in previous studies of the WCP, this study utilizes active case ascertainment methodology employed by a multidisciplinary field research team and pediatric geneticists/dysmorphologists. However, the four small towns and their surrounding rural areas studied here had never been studied for FASD epidemiology before. And they are more rural, remote, and lower SES than the community of previous WCP studies.
Section snippets
Sampling and recruitment
Active, written consent for children to participate in the study was sought from parents and guardians of all first grade pupils (n = 1866) enrolled in all 53 primary schools of the four town region and child assent forms were obtained from all children seven years and older. Consent was received for 1354 (72.6%). As in Fig. 1, a three-tier process of screening, data collection, and diagnosis was instituted for all consented children. In Tier I all children were measured for height, weight, and
Child physical growth and development and characteristic dysmorphology
In Table 1, mean values of study children for each salient demographic and growth variables and the three cardinal facial features for FASD are significantly different across the FASD continuum and the normal control group. Children with FASD were significantly smaller than controls on height, weight, BMI, OFC, and cardinal features of FAS: palpebral fissure length (PFL), smooth philtrum, and narrow vermillion. Age was also different among groups in the school sample; the FASD children were
Summary findings in these rural communities
The physical, cognitive, and behavioral traits of the children with FASD and the controls in highly rural, low SES communities of the WCP are similar to those found in other population-based studies of FASD in the WCP, yet the traits are more prevalent and severe and the prevalence of FASD higher. Child physical variables of growth and development and many minor anomalies in this sample were clearly categorized in a linear fashion by the diagnostic categories of the revised IOM criteria. No
Conclusions
The rate of FASD in these four towns and surrounding rural areas is the highest ever documented in a general population study. A historical and common pattern of recreational drinking on the weekends, coupled with poverty and low SES conditions, combine for a breathtakingly high rate of FASD. The traits of children with FAS, PFAS, and ARND are clearly different from one another and from normal controls on most every child physical, developmental, and behavioral trait, and on multiple maternal
Role of the funding source
Funding was provided by the National Institutes of Health, National Institute on Alcohol Abuse and Alcoholism (NIAAA) (UO1 AA15134).
Conflicts of interest
None of the authors have any conflicts of interest to declare.
Contributors
Philip May was the principle investigator of the NIH grant that funded this research and he, in collaboration with Julie Hasken and Barbara Tabachnick on final data analysis and Table preparation, was the major writer and final editor of all drafts. Anna-Susan Marais was the program manager who supervised all data and protocols in the main office at the Faculty of Medicine and Health Sciences of Stellenbosch University. Marlene de Vries, one of the program officers in the field office of the
Acknowledgements
We thank all of the parents who gave their permission for their children to participate in the study, the Department of Education and teachers who approved the study and supported us and the children, parents who consented their children into the study, children who participated in the study at each of the schools, and all of the mothers who provided information for the maternal risk portion this study. We are especially indebted to the fantastic field work staff for this study. Alitha Pithey
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