Unique Facial Features Distinguish Fetal Alcohol Syndrome Patients and Controls in Diverse Ethnic Populations

Authors

  • Elizabeth S. Moore,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Richard E. Ward,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Leah Flury Wetherill,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Jeffrey L. Rogers,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Ilona Autti-Rämö,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Åse Fagerlund,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Sandra W. Jacobson,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Luther K. Robinson,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • H. Eugene Hoyme,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Sarah N. Mattson,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • Tatiana Foroud,

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • the CIFASD

    1. From St. Vincent Hospital, Indianapolis, Indiana (ESM); Indiana University School of Liberal Arts, Indianapolis, Indiana (REW); Indiana University School of Medicine, Indianapolis, Indiana (LFW, TF); Indiana University School of Informatics, Indianapolis, Indiana (JLR); Department of Child Neurology, HUCH Hospital for Children and Adolescents, Helsinki, Finland; and Finnish Office for Health Technology Assessment/STAKES (IA-R); Folkhälsan Research Center, Helsinki, Finland; and Psychology Department, Åbo Akademi University, Turku, Finland (AF); Department of Psychiatry and Behavioral Neurosciences, Wayne State University School of Medicine, Detroit, Michigan (SWJ); State University of New York at Buffalo School of Medicine and Biomedical Sciences, Buffalo, New York (LKR); Stanford University School of Medicine, Stanford, California (EH); and Center for Behavioral Teratology, Department of Psychology, San Diego State University, San Diego, California (SNM).
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  • *

    The Collaborative Initiative on Fetal Alcohol Spectrum Disorders (CIFASD) (PI: E. Riley, San Diego State University) includes 8 different centers where data collection and analysis take place. The 4 sites and associated investigators that contributed data for the current project were the Folkhälsan Research Center-Helsinki (I. Autti-Rämö, Å. Fagerlund, M. Korkman), Cape Town (S. W. Jacobson), State University of New York at Buffalo School of Medicine and Biomedical Sciences (L. K. Robinson), and San Diego State University (S.N. Mattson).

  • Sources of support: This international collaborative study was supported by grants from the National Institute on Alcohol Abuse and Alcoholism (NIAAA): U24AA014809 (to T.F.), U01AA014790 (to S.J.), U24AA014815 (to K.J.), U01AA014786 (to P.M.), U01AA014834 (to S.M.), U24AA014828 (to E.R.), and U24AA014830 (to E.R.), and an administrative supplement to RO1AA09524 (to S.J.). Additional funds were provided by the National Institutes of Health Office of Research on Minority Health, the Indiana Genomics Initiative (INGEN), and the Joseph Young, Sr., Fund from the State of Michigan (to S.J.).

Reprint requests: Elizabeth Moore, PhD, 8081 Township Line Road, Suite 200, Indianapolis, IN 46260; Fax: (317) 415-8071; E-mail: esmoore@stvincent.org

Abstract

Background:  Effective management of fetal alcohol spectrum disorders (FASD) is dependent on the timely and reliable diagnosis of affected individuals. There are significant diagnostic difficulties because of the reduced prominence of facial features as children age to adulthood as well as potential population or ethnic differences in the most characteristic alcohol-related facial features.

Methods:  A total of 276 subjects were recruited from 4 sites (Cape Town, South Africa; Helsinki, Finland; Buffalo, New York; and San Diego, California) and completed a detailed dysmorphology evaluation to classify subjects as either fetal alcohol syndrome (FAS; 43%) or control (57%). Computerized anthropometry was employed to identify facial features that could distinguish FAS patients from controls across a wide age range and across ethnically disparate study populations.

Results:  Subjects were placed into 1 of 4 populations based on their ancestry (Cape Coloured, Finnish Caucasian, African American, or North American Caucasian). Analyses performed in each of the 4 study populations were able to identify a unique set of variables which provided excellent discrimination between the 2 groups (FAS, control). In each study group, at least one ocular-related measurement, shortened palpebral fissure, reduced outer canthal width, or reduced inner canthal width, was included in the final classification model.

Conclusions:  We found measurements that reflected reduced size of the eye orbit to be a consistent feature discriminating FAS and controls across each study population. However, each population had a unique, though often overlapping, set of variables which discriminated the 2 groups, suggesting important ethnic differences in the presentation of FAS. It is possible that these differences were accentuated by the wide age distribution of the study subjects.

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