Pediatrics 134(5): 855-866

PMC: PMC4210790

PMID: 25349310

Prevalence and Characteristics of Fetal Alcohol Spectrum Disorders

Julie Hasken+5 authors

OBJECTIVES:

To determine the prevalence and characteristics of fetal alcohol spectrum disorders (FASD) among first grade students (6- to 7-year-olds) in a representative Midwestern US community.

METHODS:

From a consented sample of 70.5% of all first graders enrolled in public and private schools, an oversample of small children (≤25th percentile on height, weight, and head circumference) and randomly selected control candidates were examined for physical growth, development, dysmorphology, cognition, and behavior. The children’s mothers were interviewed for maternal risk.

RESULTS:

Total dysmorphology scores differentiate significantly fetal alcohol syndrome (FAS) and partial FAS (PFAS) from one another and from unexposed controls. Alcohol-related neurodevelopmental disorder (ARND) is not as clearly differentiated from controls. Children who had FASD performed, on average, significantly worse on 7 cognitive and behavioral tests and measures. The most predictive maternal risk variables in this community are late recognition of pregnancy, quantity of alcoholic drinks consumed 3 months before pregnancy, and quantity of drinking reported for the index child’s father. From the final multidisciplinary case findings, 3 techniques were used to estimate prevalence. FAS in this community likely ranges from 6 to 9 per 1000 children (midpoint, 7.5), PFAS from 11 to 17 per 1000 children (midpoint, 14), and the total rate of FASD is estimated at 24 to 48 per 1000 children, or 2.4% to 4.8% (midpoint, 3.6%).

CONCLUSIONS:

Children who have FASD are more prevalent among first graders in this Midwestern city than predicted by previous, popular estimates.

^{Department of Nutrition, Gillings School of Global Public Health, Nutrition Research Institute, University of North Carolina at Chapel Hill, Chapel Hill, North Carolina;}

^{Center on Alcoholism, Substance Abuse and Addictions (CASAA), The University of New Mexico, Albuquerque, New Mexico;}

^{Sanford Research, Sioux Falls, South Dakota;}

^{Department of Pediatrics, University of Mississippi, Jackson, Mississippi;}

^{Department of Pediatrics, University of Washington, Seattle, Washington;}

^{Dysmorphology and Clinical Genetics, State University of New York at Buffalo, Buffalo, New York;}

^{Departments of Pathology and Pediatrics, Stanford University, Stanford, California; and}

^{Department of Pediatrics, Sanford School of Medicine, The University of South Dakota, Sioux Falls, South Dakota}

^{Corresponding author.}

Address correspondence to Philip A. May, University of North Carolina at Chapel Hill, Nutrition Research Institute, 500 Laureate Way, Kannapolis, NC 28081. E-mail: ude.cnu@yam_pilihp

Accepted 2014 Aug 27.

Abstract

OBJECTIVES:

To determine the prevalence and characteristics of fetal alcohol spectrum disorders (FASD) among first grade students (6- to 7-year-olds) in a representative Midwestern US community.

METHODS:

RESULTS:

CONCLUSIONS:

Children who have FASD are more prevalent among first graders in this Midwestern city than predicted by previous, popular estimates.

Keywords: fetal alcohol spectrum disorders, alcohol use and abuse, women, prenatal alcohol use, prevalence, children with FASD

Abstract

What’s Known on This Subject:

Most studies of fetal alcohol syndrome and fetal alcohol spectrum disorders (FASD) prevalence in the general population of the United States have been carried out using passive methods (surveillance or clinic-based studies), which underestimate rates of FASD.

What This Study Adds:

Using active case ascertainment methods among children in a representative middle class community, rates of fetal alcohol syndrome and total FASD are found to be substantially higher than most often cited estimates for the general US population.

Determining the prevalence of fetal alcohol spectrum disorders (FASD) in a general population has proved to be an elusive task. Since the diagnosis of fetal alcohol syndrome (FAS) was first described in 1973,¹ surveillance systems, prenatal clinic-based studies, and special referral clinics have proven inadequate for determining the prevalence of FAS or FASD. The often cited estimates for general populations are believed to be underestimates; yet very high rates have been found in certain substrate populations.²3 Rates from high-risk subgroups cannot be extrapolated accurately to general populations.⁴5

The Centers for Disease Control and Prevention (CDC) has estimated that FAS occurs at a rate of 0.2 to 1.5 per 1000 children,⁶7 and the Institute of Medicine (IOM) estimates are 0.5 to 3.0 per 1000 children.⁸ More current estimates of the prevalence of FAS in the US general population range from 0.2 to 7 per 1000 children,⁵ and 2% to 5% for the entire continuum of FASD.⁵9

One approach used successfully to determine the minimal prevalence of FASD in communities in South Africa,¹⁰14 Italy,¹⁵16 and Croatia¹⁷18 uses active case ascertainment in schools. Providing targeted physical examinations and cognitive/behavioral testing to primary school children¹⁹22 and interviewing their mothers²³25 can be effective for studying FASD prevalence and characteristics.

The Study Community

This study examined the prevalence and characteristics of FASD among first grade children in a representative Midwestern US city. Maternal risk factors for FASD were also explored. A total of 160 000 persons reside in the study community, among whom 87% are white. The residents are predominately middle class, with a per capita income of $28 000 and median household income of $51 800; 11% are below the poverty level. These indicators and others are virtually identical to US averages, except that US norms indicate that 14% of the general population is below the poverty level and reflect more racial diversity than the study community.²⁶ Per capita alcohol consumption in this state was 9.9 L of ethanol per year in 2009, 14% higher than the US average of 8.7 L,²⁷ but this county had an alcohol-related mortality index 27% less than the state as a whole.²⁸ The United Health Foundation²⁹ overall health ranking of this state is between 20 and 25 of 50 states. Data from the CDC Behavioral Risk Factor Surveillance System ranks the general health status of this county at 3.6 (above average) of a possible 5, and cites smoking at the US average.³⁰ In 2011, CDC data reported that 54% of females there consumed alcohol in the past 30 days, slightly higher than the US average.³¹

PFL, palpebral fissure length; —, data not collected for the whole sample on these variables.

ICD, inner canthal distance; IPD, inter-pupillary distance.

DAS, Differential Ability Scales.

Acknowledgments

The project was started with supplemental stimulus funding to the first referenced grant and was later included in the NIAAA-funded initiative, Collaborative on Fetal Alcohol Syndrome Prevalence (CoFASP) via the second referenced grant. Marcia Scott, PhD, Kenneth Warren, PhD, Faye Calhoun, DPA, and T-K Li, MD of NIAAA have provided intellectual guidance and support for prevalence studies of FASD for many years. Our deepest thanks are extended to the superintendent of schools, administrators, principals, psychologists, and teachers of the school system in the study community who have hosted and assisted in the research process over the years. Their professional support, guidance, and facilitation have been vital to the success of this study. We dedicate this paper to them and also to our deceased colleague, Dr Jason Blankenship, who invested much effort into this project before his untimely death.

Acknowledgments

Glossary

ARND	alcohol-related neurodevelopmental disorder
CDC	Centers for Disease Control and Prevention
CI	95% confidence interval
FASD	fetal alcohol spectrum disorders
FAS	fetal alcohol syndrome
IOM	Institute of Medicine
OFC	occipitofrontal (head) circumference
PFAS	partial fetal alcohol syndrome
SES	socioeconomic status

Glossary

Footnotes

^Deceased.

Dr May is the Principal Investigator who designed the study, received the NIH funding, and wrote the majority of the first and last drafts of the manuscript; Ms Baete, Mr Russo, and Dr Elliott were respectively the program manager, project officer, and co-investigator who organized, implemented, and administered much of the data collection in the Midwestern city, and each contributed written text and edited various drafts of the manuscript; Dr Blankenship performed much of the data and statistical analyses, contributed written text and interpretation, constructed some tables, and edited various drafts of the manuscript before his sudden death on October 29, 2013; Ms Kalberg, Mr Buckley, and Ms Brooks were the designers and supervisors of the field data collection, data management, data entry, and Institutional Review Board activities, and each read drafts and edited the manuscript; Ms Hasken contributed to and edited each draft of the manuscript and produced original figures and the final tables and figures; Drs Abdul-Rahman, Adam, Robinson, and Manning were project dysmorphologists who diagnosed and generated clinical dysmorphology data in field clinics and made final diagnoses; Dr Hoyme was co-Principal Investigator for the site and the chief dysmorphologist supervising the clinical team members, administering clinical examinations, and generating data, he was chief medical officer assuming responsibility for medical liaison with the schools, families, and local administration, and he contributed written material and edited various drafts of the manuscript; and all authors approved the final manuscript as submitted.

FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.

FUNDING: This project was funded by the National Institutes of Health (NIH), the National Institute on Alcohol Abuse, and Alcoholism (NIAAA), grants R01 AA11685, and RO1/UO1 AA01115134. Funded by the National Institutes of Health (NIH).

POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose.

Footnotes

References

1. Jones KL, Smith DW. Recognition of the fetal alcohol syndrome in early infancy.Lancet. 1973;302(7836):999–1001 [[PubMed]
2. Robinson GC, Conry JL, Conry RF. Clinical profile and prevalence of fetal alcohol syndrome in an isolated community in British Columbia.CMAJ. 1987;137(3):203–207
3. May PA, Hymbaugh KJ, Aase JM, Samet JM. Epidemiology of fetal alcohol syndrome among American Indians of the Southwest.Soc Biol. 1983;30(4):374–387 [[PubMed]
4. May PA, Gossage JP. Estimating the prevalence of fetal alcohol syndrome. A summary.Alcohol Res Health. 2001;25(3):159–167
5. May PA, Gossage JP, Kalberg WO, et al. . Prevalence and epidemiologic characteristics of FASD from various research methods with an emphasis on recent in-school studies.Dev Disabil Res Rev. 2009;15(3):176–192 [[PubMed]
6. Centers for Disease Control and Prevention (CDC) . Surveillance for fetal alcohol syndrome using multiple sources — Atlanta, Georgia, 1981-1989.MMWR Morb Mortal Wkly Rep. 1997;46(47):1118–1120 [[PubMed]
7. Centers for Disease Control and Prevention (CDC) . Update: trends in fetal alcohol syndrome—United States, 1979-1993.MMWR Morb Mortal Wkly Rep. 1995;44(13):249–251 [[PubMed]
8. Stratton KR, Howe CJ, Battaglia FC, editors, eds. Fetal Alcohol Syndrome Diagnosis, Epidemiology, Prevention, and Treatment. Washington, DC: National Academy Press; 1996 [PubMed][Google Scholar]
9. Sampson PD, Streissguth AP, Bookstein FL, et al. . Incidence of fetal alcohol syndrome and prevalence of alcohol-related neurodevelopmental disorder.Teratology. 1997;56(5):317–326 [[PubMed]
10. May PA, Brooke L, Gossage JP, et al. . Epidemiology of fetal alcohol syndrome in a South African community in the Western Cape Province.Am J Public Health. 2000;90(12):1905–1912
11. May PA, Gossage JP, Marais AS, et al. . The epidemiology of fetal alcohol syndrome and partial FAS in a South African community.Drug Alcohol Depend. 2007;88(2-3):259–271
12. May PA, Blankenship J, Marais AS, et al. . Approaching the prevalence of the full spectrum of fetal alcohol spectrum disorders in a South African population-based study.Alcohol Clin Exp Res. 2013;37(5):818–830
13. Viljoen DL, Gossage JP, Brooke L, et al. . Fetal alcohol syndrome epidemiology in a South African community: a second study of a very high prevalence area.J Stud Alcohol. 2005;66(5):593–604
14. Urban M, Chersich MF, Fourie LA, Chetty C, Olivier L, Viljoen D. Fetal alcohol syndrome among grade 1 schoolchildren in Northern Cape Province: prevalence and risk factors.S Afr Med J. 2008;98(11):877–882 [[PubMed]
15. May PA, Fiorentino D, Phillip Gossage J, et al. . Epidemiology of FASD in a province in Italy: Prevalence and characteristics of children in a random sample of schools.Alcohol Clin Exp Res. 2006;30(9):1562–1575 [[PubMed]
16. May PA, Fiorentino D, Coraile G, et al. . Prevalence of children with fetal alcohol spectrum disorders in communities near Rome, Italy: rates are substantially higher than previous estimates.Int J Environ Res Public Health. 2011;8(6):2331–2351
17. Petković G, Barisić I. FAS prevalence in a sample of urban schoolchildren in Croatia.Reprod Toxicol. 2010;29(2):237–241 [[PubMed]
18. Petković G, Barišić I. Prevalence of fetal alcohol syndrome and maternal characteristics in a sample of schoolchildren from a rural province of Croatia.Int J Environ Res Public Health. 2013;10(4):1547–1561
19. Adnams CM, Kodituwakku PW, Hay A, Molteno CD, Viljoen D, May PA. Patterns of cognitive-motor development in children with fetal alcohol syndrome from a community in South Africa.Alcohol Clin Exp Res. 2001;25(4):557–562 [[PubMed]
20. Kodituwakku P, Coriale G, Fiorentino D, et al. . Neurobehavioral characteristics of children with fetal alcohol spectrum disorders in communities from Italy: Preliminary results.Alcohol Clin Exp Res. 2006;30(9):1551–1561 [[PubMed]
21. Aragón AS, Coriale G, Fiorentino D, et al. . Neuropsychological characteristics of Italian children with fetal alcohol spectrum disorders.Alcohol Clin Exp Res. 2008;32(11):1909–1919
22. Kalberg WO, May PA, Blankenship J, Buckley D, Gossage JP, Adnams CM. A practical testing battery to measure neurobehavioral ability among children with FASD.Int J Alcohol Drug Res. 2013;2(3):51–60
23. Viljoen D, Croxford J, Gossage JP, Kodituwakku PW, May PA. Characteristics of mothers of children with fetal alcohol syndrome in the Western Cape Province of South Africa: a case control study.J Stud Alcohol. 2002;63(1):6–17 [[PubMed]
24. May PA, Gossage JP, Brooke LE, et al. . Maternal risk factors for fetal alcohol syndrome in the Western cape province of South Africa: a population-based study.Am J Public Health. 2005;95(7):1190–1199
25. May PA, Gossage JP, Marais AS, et al. . Maternal risk factors for fetal alcohol syndrome and partial fetal alcohol syndrome in South Africa: a third study.Alcohol Clin Exp Res. 2008;32(5):738–753 [[PubMed]
26. US Census Bureau. State & County Quick Facts. Available at: . Accessed April 1, 2014[PubMed]
27. LaVallee RA, Yi H. Surveillance Report #92: Apparent Per Capita Alcohol Consumption: National, State, And Regional Trends, 1977–2009. Rockville, MD: National Institute on Alcohol Abuse and Alcoholism; 2011 [PubMed]
28. US Department of Health and Human Service. County Alcohol Problem Indicators 1989–1990. Rockville, MD; 1994 [PubMed]
29. United Health Foundation. America’s Health Rankings. Available at: . Accessed April 1, 2014[PubMed]
30. US countiesAvailable at: . Accessed April 1, 2014[PubMed]
31. Office of Health Statistics The Health Behaviors of South Dakotans 2011. Pierre, SD: Centers for Disease Control and Prevention; 2013 [PubMed]
32. Hoyme HE, May PA, Kalberg WO, et al. . A practical clinical approach to diagnosis of fetal alcohol spectrum disorders: clarification of the 1996 institute of medicine criteria.Pediatrics. 2005;115(1):39–47
33. Alvik A, Haldorsen T, Lindemann R. Alcohol consumption, smoking and breastfeeding in the first six months after delivery.Acta Paediatr. 2006;95(6):686–693 [[PubMed]
34. May PA, Tabachnick BG, Gossage JP, et al. . Maternal risk factors predicting child physical characteristics and dysmorphology in fetal alcohol syndrome and partial fetal alcohol syndrome.Drug Alcohol Depend. 2011;119(1-2):18–27
35. Wurst FM, Kelso E, Weinmann W, Pragst F, Yegles M, Sundström Poromaa, IMeasurement of direct ethanol metabolites suggests higher rate of alcohol use among pregnant women than found with the AUDIT—a pilot study in a population-based sample of Swedish women. Am J Obstet Gynecol. 2008;198(4):407.e1–e5 [[PubMed]
36. Ervalahti N, Korkman M, Fagerlund A, Autti-Rämö I, Loimu L, Hoyme HE. Relationship between dysmorphic features and general cognitive function in children with fetal alcohol spectrum disorders.Am J Med Genet A. 2007;143A(24):2916–2923 [[PubMed]
37. Beery K, Buktenica N, Beery N. Beery-Buktenica Developmental Test of Visual-Motor Integration, 5th ed. San Antonio, TX: Pearson Assessments; 2006 [PubMed]
38. Elliott CD. Differential Ability Scales, 2nd ed. San Antonio, TX: Harcourt Assessment, Inc, Pearson Education, Inc; 2007 [PubMed]
39. Sparrow SS, Cicchetti DV, Balla DA. Vineland Adaptive Behavior Scales, 2nd ed. San Antonio, TX: Pearson Assessments; 2006 [PubMed]
40. Sobell LC, Agrawal S, Annis H, et al. . Cross-cultural evaluation of two drinking assessment instruments: alcohol timeline followback and inventory of drinking situations.Subst Use Misuse. 2001;36(3):313–331 [[PubMed]
41. Sobell LC, Sobell MB, Leo GI, Cancilla A. Reliability of a timeline method: assessing normal drinkers’ reports of recent drinking and a comparative evaluation across several populations.Br J Addict. 1988;83(4):393–402 [[PubMed]
42. Kaskutas LA, Graves K. An alternative to standard drinks as a measure of alcohol consumption.J Subst Abuse. 2000;12(1-2):67–78 [[PubMed]
43. Kaskutas LA, Graves K. Pre-pregnancy drinking: how drink size affects risk assessment.Addiction. 2001;96(8):1199–1209 [[PubMed]
44. Kaskutas LA, Kerr WC. Accuracy of photographs to capture respondent-defined drink size.J Stud Alcohol Drugs. 2008;69(4):605–610 [[PubMed]
45. King AC. Enhancing the self-report of alcohol consumption in the community: two questionnaire formats.Am J Public Health. 1994;84(2):294–296
46. Alvik A, Haldorsen T, Groholt B, Lindemann R. Alcohol consumption before and during pregnancy comparing concurrent and retrospective reports.Alcohol Clin Exp Res. 2006;30(3):510–515 [[PubMed]
47. Czarnecki DM, Russell M, Cooper ML, Salter D. Five-year reliability of self-reported alcohol consumption.J Stud Alcohol. 1990;51(1):68–76 [[PubMed]
48. Hannigan JH, Chiodo LM, Sokol RJ, et al. . A 14-year retrospective maternal report of alcohol consumption in pregnancy predicts pregnancy and teen outcomes.Alcohol. 2010;44(7-8):583–594
49. Microsoft Excel [computer software]. Version 2007. Redmond, WA: Microsoft; 2007
50. IBM SPSS Statistics for Windows [computer program]. Version 20.0. Armonk, NY: IBM Corp; 2011
51. Tabachnick BG, Fidell LS. Using Multivariate Statistics, 6th ed. Boston, MA: Pearson; 2013 [PubMed]
52. May PA, Gossage JP. Maternal risk factors for fetal alcohol spectrum disorders: not as simple as it might seem.Alcohol Res Health. 2011;34(1):15–26
53. Feldman HS, Jones KL, Lindsay S, et al. . Prenatal alcohol exposure patterns and alcohol-related birth defects and growth deficiencies: a prospective study.Alcohol Clin Exp Res. 2012;36(4):670–676 [[PubMed]
54. May PA, Tabachnick BG, Gossage JP, et al. . Maternal factors predicting cognitive and behavioral characteristics of children with fetal alcohol spectrum disorders.J Dev Behav Pediatr. 2013;34(5):314–325
55. Abel EL. Fetal Alcohol Abuse Syndrome. New York, NY: Plenum Press; 1998 [PubMed]
56. Balachova T, Bonner B, Chaffin M, et al. . Women’s alcohol consumption and risk for alcohol-exposed pregnancies in Russia.Addiction. 2012;107(1):109–117
57. Floyd RL, Decouflé P, Hungerford DW. Alcohol use prior to pregnancy recognition.Am J Prev Med. 1999;17(2):101–107 [[PubMed]
58. Mallard SR, Connor JL, Houghton LA. Maternal factors associated with heavy periconceptional alcohol intake and drinking following pregnancy recognition: a post-partum survey of New Zealand women.Drug Alcohol Rev. 2013;32(4):389–397 [[PubMed]
59. Parackal SM, Parackal MK, Harraway JA. Prevalence and correlates of drinking in early pregnancy among women who stopped drinking on pregnancy recognition.Matern Child Health J. 2013;17(3):520–529 [[PubMed]
60. Russell M, Czarnecki DM, Cowan R, McPherson E, Mudar PJ. Measures of maternal alcohol use as predictors of development in early childhood.Alcohol Clin Exp Res. 1991;15(6):991–1000 [[PubMed]
61. Skagerström J, Alehagen S, Häggström-Nordin E, Årestedt K, Nilsen P. Prevalence of alcohol use before and during pregnancy and predictors of drinking during pregnancy: a cross sectional study in Sweden.BMC Public Health. 2013;13:780.
62. Alvik A, Haldorsen T, Lindemann R. Consistency of reported alcohol use by pregnant women: anonymous versus confidential questionnaires with item nonresponse differences.Alcohol Clin Exp Res. 2005;29(8):1444–1449 [[PubMed]
63. Alvik A, Heyerdahl S, Haldorsen T, Lindemann R. Alcohol use before and during pregnancy: a population-based study.Acta Obstet Gynecol Scand. 2006;85(11):1292–1298 [[PubMed]
64. Ortega-García JA, Gutierrez-Churango JE, Sánchez-Sauco MF, et al. . Head circumference at birth and exposure to tobacco, alcohol and illegal drugs during early pregnancy.Childs Nerv Syst. 2012;28(3):433–439
65. Bryanton J, Gareri J, Boswall D, et al. . Incidence of prenatal alcohol exposure in Prince Edward Island: a population-based descriptive study.CMAJ Open. 2014;2(2):E121–E126
66. Manich A, Velasco M, Joya X, et al. . [Validity of a maternal alcohol consumption questionnaire in detecting prenatal exposure]. An Pediatr (Barc). 2012;76(6):324–328 [[PubMed]
67. Morini L, Marchei E, Tarani L, et al. . Testing ethylglucuronide in maternal hair and nails for the assessment of fetal exposure to alcohol: comparison with meconium testing.Ther Drug Monit. 2013;35(3):402–407 [[PubMed]
68. Pichini S, Marchei E, Vagnarelli F, et al. . Assessment of prenatal exposure to ethanol by meconium analysis: results of an Italian multicenter study.Alcohol Clin Exp Res. 2012;36(3):417–424 [[PubMed]