RT Journal Article SR Electronic T1 Strain-Dependent Modifier Genes Determine Survival in Zfp423 Mice JF G3: Genes|Genomes|Genetics FD Genetics Society of America SP 4241 OP 4247 DO 10.1534/g3.120.401720 VO 10 IS 11 A1 Alcaraz, Wendy A. A1 Liu, Zheng A1 Valdes, Phoebe A1 Chen, Edward A1 Valdovino Gonzalez, Alan G. A1 Wade, Shelby A1 Wong, Cinny A1 Kim, Eunnie A1 Chen, Hsiang-Hua M. A1 Ponn, Alison A1 Concepcion, Dorothy A1 Hamilton, Bruce A. YR 2020 UL http://www.g3journal.org/content/10/11/4241.abstract AB Zfp423 encodes a transcriptional regulatory protein that interacts with canonical signaling and lineage pathways. Mutations in mouse Zfp423 or its human ortholog ZNF423 are associated with a range of developmental abnormalities reminiscent of ciliopathies, including cerebellar vermis hypoplasia and other midline brain defects. Null mice have reduced viability in most strain backgrounds. Here we show complete lethality on a C57BL/6J background, dominant rescue in backcrosses to any of 13 partner strains, with strain-dependent survival frequencies, and evidence for a BALB/c-derived survival modifier locus on chromosome 5. Survival data indicate both perinatal and postnatal periods of lethality. Anatomical data from a hypomorphic gene trap allele observed on both C57BL/6J and BALB/c congenic backgrounds shows an aggregate effect of background on sensitivity to Zfp423 loss rather than a binary effect on viability.