Abstract
Genetic factors influence the physical and neurobehavioral manifestations of prenatal alcohol exposure (PAE). Animal models allow the investigation of specific genes that confer vulnerability to, or protection from, birth defects associated with fetal alcohol spectrum disorders (FASDs). The objective of the present experiments was to determine if genetic alterations in the Sonic Hedgehog (Shh) signaling pathways affect the vulnerability to PAE-induced skeletal defects involving the forelimbs and/or hindlimbs. Wild-type C57BL/6J female mice were bred with males in which one copy of the Shh or Gli2 genes had been knocked out, to produce litters with both wild-type (+/+) and heterozygous (+/-) embryos. Alcohol doses (two injections of 2.9 g/kg, 4 hours apart) or vehicles were administered starting at gestational day (GD) 9.25, 9.5, or 9.75, a critical exposure time for inducing limb defects. Limb defects were examined at GD 17 using a dysmorphology scale based on abnormalities ranging from increased interdigital spacing to the deletion of multiple fingers and the ulna. Alcohol treatment caused a high incidence of forelimb defects, particularly on the right side, that was higher in Shh+/- and Gli2+/- fetuses compared to wild-type f...Continue Reading
References
Jun 1, 1977·Teratology·G F Chernoff
Oct 1, 1992·Teratology·L E KotchK K Sulik
Jun 1, 1991·Alcoholism, Clinical and Experimental Research·G Duester
Apr 1, 1990·Teratology·E F ZimmermanM D Collins
Jun 1, 1986·Teratology·R M Pauli, P F Feldman
Dec 1, 1980·Teratology·M J McLeod
May 1, 1980·European Journal of Pediatrics·J HerrmannJ M Opitz
Jul 1, 1997·Alcohol·S L BoehmD M Gilliam
Jul 26, 2002·Proceedings of the National Academy of Sciences of the United States of America·Sara C AhlgrenMarianne Bronner-Fraser
Aug 29, 2002·Nature·Ying LitingtungChin Chiang
Apr 27, 2004·Birth Defects Research. Part A, Clinical and Molecular Teratology·Kimberly ChrismanDavid P Gardner
Nov 19, 2004·Developmental Psychobiology·Andrew P McMechanJohn H Hannigan
Jan 24, 2007·Laboratory Investigation; a Journal of Technical Methods and Pathology·Yin-Xiong LiAnna Mae Diehl
Aug 7, 2007·Birth Defects Research. Part A, Clinical and Molecular Teratology·Corey S JohnsonKathleen K Sulik
Dec 24, 2008·Developmental Biology·Kazushi AotoJun Motoyama
Nov 19, 2009·Nature Reviews. Genetics·Rolf ZellerAimée Zuniga
May 1, 2010·Journal of Developmental and Behavioral Pediatrics : JDBP·Philip A MayH Eugene Hoyme
Oct 17, 2012·PLoS Genetics·Mingi Hong, Robert S Krauss
Jun 27, 2013·Wiley Interdisciplinary Reviews. Developmental Biology·Aimée ZunigaSimone Probst
Mar 4, 2014·PloS One·Henry W KietzmanRobert J Lipinski
Aug 19, 2014·Birth Defects Research. Part A, Clinical and Molecular Teratology·Kristin M Caspers ConwayUNKNOWN National Birth Defects Prevention Study
Aug 26, 2014·Frontiers in Genetics·Susan M SmithGeorge R Flentke
Mar 13, 2016·Current Topics in Developmental Biology·Robert S Krauss, Mingi Hong
Apr 29, 2016·Alcoholism, Clinical and Experimental Research·Johann K Eberhart, Scott E Parnell
Citations
Sep 12, 2018·Journal of Pediatric Orthopedics. Part B·Janet L WalkerScott A Riley
Mar 16, 2019·Epigenomics·Jan Maarten CobbenMarcel Mam Mannens
Feb 13, 2019·Alcoholism, Clinical and Experimental Research·Michael W Miller
Apr 30, 2019·Birth Defects Research·Shama BhatiaPeter G Wells
May 31, 2020·Alcoholism, Clinical and Experimental Research·Oswald Boa-AmponsemGregory J Cole
Jun 13, 2018·Molecular Psychiatry·Friederike EhrhartLeopold M G Curfs
Jun 7, 2018·G3 : Genes - Genomes - Genetics·Tatiana V MorozovaRobert R H Anholt
Dec 6, 2020·Alcoholism, Clinical and Experimental Research·Julie A KableUNKNOWN CIFASD
Jun 18, 2021·Disease Models & Mechanisms·Karen E BoschenScott E Parnell
Dec 4, 2021·Birth Defects Research·Drayton C HarveyS Ram Kumar
Jul 4, 2021·NPJ Genomic Medicine·Zhigang WangWenjun Shang