TY - JOUR
T1 - Impact of genetic background on neonatal lethality of Gga2 gene-trap mice
AU - Doray, Balraj
AU - Govero, Jennifer
AU - Kornfeld, Stuart
PY - 2014
Y1 - 2014
N2 - The functional redundancy of the three mammalian Golgi-localized, α-ear-containing, ADPribosylation factor-binding proteins (GGAs) was addressed in a previous study. Using insertional mutagenesis, we found that Gga1 or Gga3 homozygous knockout mice were for the most part normal, whereas mice homozygous for two different Gga2 gene-trap alleles exhibited either embryonic or neonatal lethality in the C57BL/6 background, depending on the source of the vector utilized (Byg vs. Tigm, respectively). We now show that the Byg strain harbors a disrupted Gga2 allele that is hypomorphic, indicating that the Byg lethality is attributable to a mechanism independent of GGA2. This is in contrast to the Tigm Gga2 allele, which is a true knockout and establishes a role for GGA2 during the neonatal period. Placement of the Tigm Gga2 allele into the C57BL6/Ola129Sv mixed background results in a lower incidence of neonatal lethality, showing the importance of genetic background in determining the requirement for GGA2 during this period. The Gga2-/- mice that survive have reduced body weight at birth and this runted phenotype is maintained through adulthood.
AB - The functional redundancy of the three mammalian Golgi-localized, α-ear-containing, ADPribosylation factor-binding proteins (GGAs) was addressed in a previous study. Using insertional mutagenesis, we found that Gga1 or Gga3 homozygous knockout mice were for the most part normal, whereas mice homozygous for two different Gga2 gene-trap alleles exhibited either embryonic or neonatal lethality in the C57BL/6 background, depending on the source of the vector utilized (Byg vs. Tigm, respectively). We now show that the Byg strain harbors a disrupted Gga2 allele that is hypomorphic, indicating that the Byg lethality is attributable to a mechanism independent of GGA2. This is in contrast to the Tigm Gga2 allele, which is a true knockout and establishes a role for GGA2 during the neonatal period. Placement of the Tigm Gga2 allele into the C57BL6/Ola129Sv mixed background results in a lower incidence of neonatal lethality, showing the importance of genetic background in determining the requirement for GGA2 during this period. The Gga2-/- mice that survive have reduced body weight at birth and this runted phenotype is maintained through adulthood.
KW - GGA2
KW - Gene-trap hypomorphic allele
KW - Genetic background
KW - Neonatal lethality
UR - http://www.scopus.com/inward/record.url?scp=84901368061&partnerID=8YFLogxK
U2 - 10.1534/g3.114.010355
DO - 10.1534/g3.114.010355
M3 - Article
C2 - 24637350
AN - SCOPUS:84901368061
SN - 2160-1836
VL - 4
SP - 885
EP - 890
JO - G3: Genes, Genomes, Genetics
JF - G3: Genes, Genomes, Genetics
IS - 5
ER -