Genetic vulnerabilities to prenatal alcohol exposure: Limb defects in sonic hedgehog and GLI2 heterozygous mice

Birth Defects Research
Eric W FishScott E Parnell

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

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Citations

Sep 12, 2018·Journal of Pediatric Orthopedics. Part B·Janet L WalkerScott A Riley
Feb 13, 2019·Alcoholism, Clinical and Experimental Research·Michael W Miller
Apr 30, 2019·Birth Defects Research·Shama BhatiaPeter G Wells
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

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Birth Defects

Birth defects encompass structural and functional alterations that occur during embryonic or fetal development and are present since birth. The cause may be genetic, environmental or unknown and can result in physical and/or mental impairment. Here is the latest research on birth defects.