Prevalence and characteristics of fetal alcohol syndrome and partial fetal alcohol syndrome in a Rocky Mountain Region City
Introduction
The rate of fetal alcohol syndrome (FAS) in the United States (US) was estimated for many years, and believed by many, to be 0.33–3.0 per 1000 children (Abel, 1998, Abel and Sokol, 1987, Abel and Sokol, 1991, CDC, 1995, CDC, 1997, Fox et al., 2015, May and Gossage, 2001; Stratton et al., 1996). However, given the lack of active case ascertainment studies (which seek to identify cases in a general population) of any fetal alcohol spectrum disorders (FASD) in the US and other developed countries (May et al., 2009), many people believed that the above estimates were substantial under estimates. FASD are rarely recognized or diagnosed in the general population or in general clinical settings (Chasnoff et al., 2015). Therefore, surveillance and clinic-based studies are unlikely to produce a true prevalence (Fox et al., 2015). Active case ascertainment studies undertaken in schools have proven to be accurate and to produce high rates of FASD (May et al., 2009). In-school study methods for large communities have been employed mostly in South Africa and Italy (May et al., 2000, May et al., 2006, May et al., 2007, May et al., 2011, May et al., 2013a, Urban et al., 2008, Viljoen et al., 2005).
Prior to 2014, only three articles had been published that utilized active case ascertainment methods in schools in the US general population. Clarren et al. (2001) reported a rate of 3.1 FAS cases per 1000 in a county in Washington State. In that study, only one of the seven FAS cases found in the schools had been diagnosed previously. Burd et al. (1999) and Poitra et al. (2003) have reported on active case ascertainment methods used in multiple years in Head Start classrooms using a screening tool and follow up dysmorphology exam. Burd et al. (1999) reported six cases of FAS out of 1013 children, 5.9 per 1000. Poitra et al. (2003) included more children from the same community and 4.3 children per 1000 had FAS. The authors do mention that some of the children with FAS identified in the Head Start studies had been diagnosed prior to the study, but this community benefited from an ongoing active dysmorphology consultation service. Many undiagnosed cases of FAS and other FASD exist in the US population, and active case ascertainment studies are designed to find them and to estimate the true prevalence of FASD.
The purpose of this study was to: (1) determine the feasibility of using these particular active case ascertainment methods in a US community to identify children with FAS and partial fetal alcohol syndrome (PFAS) in public and private schools; (2) ascertain maternal risk factors for FASD in a general US population; and (3) determine the prevalence and characteristics of children with FAS and PFAS in this community. Data were collected in three samples of first grade students in this Rocky Mountain Region City (RMRC). This study was initiated at the invitation of the city/county health department, endorsed by school administrators, and approved by the Board of Trustees of the City Schools. The studies utilized research methods pioneered in South Africa and Italy.
The population of RMRC is 59,000 in a county of 85,000. The composition of the population is 88.5% White, 5.0% American Indian, 3.8% mixed race, 1.1% Black, and 3.4% of Hispanic ethnicity (US Census, 2015). Compared to the US population, the study community was more White (+10.8%), less Black (−12.1%), more American Indian (+3.8%) and less Hispanic (−13.7%). RMRC residents are predominantly middle class, with average economic indicators similar to small cities and counties in the region and state. The averages in the county are slightly below US averages on a number of economic indicators: per capita income is $24,100 (US average is $28,155), median household income is $43,800 (US = $53,000), and 16.5% are below the poverty level (US = 15.4%) (US Census, 2015). Per capita alcohol consumption in this state was 2.99 gallons (11.3 l) of ethanol per year in 2009 compared to the US average of 2.3 gallons (8.7 l) (LaVallee and Yi, 2011). The overall health rank of this state by the United Health Foundation (2014) is well in the upper middle tier, between 20 and 25 of 50 states. County data from the CDC Behavioral Risk Factor Surveillance System (BRFSS) indicates less binge drinking overall (13.3%) than in either the state (16.9%) or the US (15.5%); however among adults ages 18–44, 18.3% binge drink which compares to 24.5% for the state (“Western” County, 2011). Heavy drinking was reported for this county at 4.9%, compared to 5.9% for the state, and 5.1% for the US (“Western” County, 2011). Among adults 18–44 years, heavy drinking was 5.6% in the County and 6.9% for the State (“Western” County, 2011). Binge drinking among women (four or more drinks per occasion) was lower at 14.3% in this state than among males (five or more drinks per occasion) at 27.3% (“Western State” BRFSS, 2014), but bingeing among women was 2.2% higher than US averages (12.5%) (CDC, 2013). Therefore, this site has slightly lower economic status than US averages, overall good health, but indicators of heavy drinking and binge drinking are lower than the home state but higher than US averages.
Section snippets
Methods
The diagnostic criteria used for FAS and PFAS (see Fig. 1) were originally set forth by the FAS advisory group of the U.S. Institute of Medicine (Stratton et al., 1996), and operationalized and updated from clinical experience (Hoyme et al., 2005). The classification of the children is based on: (1) specific facial dysmorphology, (2) diminished physical stature and/or weight, (3) defined intellectual, developmental, social, and neuropsychological assessments, and (4) multiple measures of
Child physical growth traits and dysmorphology
Table 1 summarizes demographic and physical features of all consented children (column 1), randomly-selected controls (column 2, n = 190), and children with final diagnoses of PFAS (n = 19), and FAS (n = 7). Values for the common measures of the randomly-selected control sample and those of consented children are similar; randomly-selected children are well within overall growth parameters. The children of the diagnostic categories did not differ significantly on sex or race/ethnicity. However, the
Discussion
Active case ascertainment methods for identifying children with lagging growth and development and significant dysmorphia associated with FAS and PFAS worked well in this population. Non-reporting or underreporting of maternal drinking, however was a greater challenge in this US community than encountered in South Africa, but comparable to the challenges in Italy and other first world countries (Alvik et al., 2005, Alvik et al., 2006, Ceccanti et al., 2014, Garcia-Algar et al., 2008, Manich et
Role of the funding source
The protocols and consent forms used in the study were approved by the IRB of the University of New Mexico. This project was funded by the National Institutes of Health (NIH), the National Institute on Alcohol Abuse, and Alcoholism (NIAAA) RO1/U01 AA11685.
Contributors
Philip May was the principle investigator (PI), and along with assistance from Julie Hasken, was the major writer and final editor of all drafts. Jason Blankenship, Wendy Kalberg, David Buckley, Jan Gossage, and Marita Brooks all played major roles in data analysis and preparation of text for the methods and results sections. Luther Robinson, Melanie Manning, and Gene Hoyme generated all dysmorphology data and made final diagnoses of the children in the field. Carol Keaster, Rosemary Bozeman,
Conflicts of interest
None of the authors have any conflicts of interest to declare.
Acknowledgements
We gratefully thank our colleagues who assisted in the study, especially the Rocky Mountain City/County health officials, the Trustees and administrators of the public and private education systems, primary school principals, school secretaries, psychologists, teachers, and others who graciously received us and facilitated the study. Craig Sivak and Mary Kay Burns also preformed maternal interviews, clinical coordination, reserved transportation, and drove the “Green Breeze”. We further
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