Document Detail


Fetal alcohol syndrome and DiGeorge anomaly: critical ethanol exposure periods for craniofacial malformations as illustrated in an animal model.
MedLine Citation:
PMID:  3146306     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Acute maternal ethanl (alcohol) administration induces different craniofacial anomalies in the offspring of experimental animals, depending on the gestational day of teratogen exposure. Previous studies in our laboratories have illustrated the sequence of developmental changes leading to the "typical" fetal alcohol syndrome (FAS) craniofacial phenotype which results from teratogen exposure during gastrulation. These facial features are accompanied by deficiencies in median forebrain derivatives. Ethanol teratogenesis at this time apparently results in a loss of midline territory of the embryonic disc with little effects on neural crest-dependent laterally derived structures including the visceral arches. Acute ethanol exposure in mice 1 1/2 days later, at a time when neural crest cells are populating the frontonasal prominence and the visceral arches, results in a craniofacial phenotype that is similar to that noted in the DiGeorge anomaly or sequence. Sequential scanning electron microscopic analysis in our laboratory of embryos exposed on day 8 1/2 have illustrated deficiencies in the developing facial prominences and the visceral arches. The developing forebrain and midbrain appear hypoplastic. We have also observed heart, great vessel, and thymus abnormalities in these fetuses. Histologic analyses indicate that a common pathogenetic basis for the above-mentioned (day 8 1/2-induced) fetal alcohol effects appears to be an interference with the integrity of the cranial (including occipital) neural crest. Other discrete cell populations may also be involved since we have observed abnormalities in other regions, including placodal and closing membrane tissues. This animal model provides evidence linking maternal ethanol abuse during the 3rd or 4th weeks of human gestation to the development in the conceptus of FAS or DiGeorge anomally craniofacial characteristics, respectively. As the DiGeorge anomaly has been noted in the offspring of alcoholic women, this animal model indicates that ethanol and/or its metabolites is, in these cases, the causative agent.
Authors:
K K Sulik; M C Johnston; P A Daft; W E Russell; D B Dehart
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.    
Journal Detail:
Title:  American journal of medical genetics. Supplement     Volume:  2     ISSN:  1040-3787     ISO Abbreviation:  Am J Med Genet Suppl     Publication Date:  1986  
Date Detail:
Created Date:  1989-03-23     Completed Date:  1989-03-23     Revised Date:  2007-11-15    
Medline Journal Info:
Nlm Unique ID:  8706133     Medline TA:  Am J Med Genet Suppl     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  97-112     Citation Subset:  IM    
Affiliation:
Department of Anatomy, School of Medicine, University of North Carolina, Chapel Hill 27514.
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MeSH Terms
Descriptor/Qualifier:
Animals
Brain / embryology,  ultrastructure
DiGeorge Syndrome / chemically induced*,  embryology,  pathology
Disease Models, Animal
Embryo, Mammalian / ultrastructure
Ethanol / toxicity*
Facial Bones / abnormalities*,  embryology,  pathology
Female
Fetal Alcohol Syndrome / embryology*,  pathology
Immunologic Deficiency Syndromes / chemically induced*
Mice
Microscopy, Electron, Scanning
Pregnancy
Skull / abnormalities*,  embryology,  pathology
Teratogens*
Grant Support
ID/Acronym/Agency:
5-501-FR-05406//PHS HHS; DE02668/DE/NIDCR NIH HHS; DE07149/DE/NIDCR NIH HHS
Chemical
Reg. No./Substance:
0/Teratogens; 64-17-5/Ethanol

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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