Dorsal surface and block face micrographs of unaffected (left), Zfp423-hypomorphic (middle), and Zfp423-null (right) mutant brains show sensitivity of midline structures to reduced Zfp423 function. Brain development requires coordination among cell lineage and extracellular signaling information. Zfp423's zinc finger domains bind DNA and other regulatory proteins in lineage- and signal-dependent contexts, including EBF, SMAD, retinoic acid receptor, notch intracellular domain, and DNA damage response proteins. Simple structural measures are sensitive to even heterozygous loss of Zfp423, allowing rapid phenotyping of variants. Among reported patient amino acid substitution variants, however, only H1277Y, which disrupts an EBF-binding zinc finger, shows an effect. See Deshpande et al.
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Image Credit: Ojas Deshpande
Research Articles
A mega-analysis of expression quantitative trait loci in retinal tissue
PLOS Genetics: published September 1, 2020 | https://doi.org/10.1371/journal.pgen.1008934
Trappc9 deficiency causes parent-of-origin dependent microcephaly and obesity
PLOS Genetics: published September 2, 2020 | https://doi.org/10.1371/journal.pgen.1008916
Trichoderma reesei XYR1 activates cellulase gene expression via interaction with the Mediator subunit TrGAL11 to recruit RNA polymerase II
PLOS Genetics: published September 2, 2020 | https://doi.org/10.1371/journal.pgen.1008979
Excess crossovers impede faithful meiotic chromosome segregation in C. elegans
PLOS Genetics: published September 4, 2020 | https://doi.org/10.1371/journal.pgen.1009001
Male-biased aganglionic megacolon in the TashT mouse model of Hirschsprung disease involves upregulation of p53 protein activity and Ddx3y gene expression
PLOS Genetics: published September 8, 2020 | https://doi.org/10.1371/journal.pgen.1009008
Genetic analysis of the modern Australian labradoodle dog breed reveals an excess of the poodle genome
PLOS Genetics: published September 10, 2020 | https://doi.org/10.1371/journal.pgen.1008956
Adiponectin GWAS loci harboring extensive allelic heterogeneity exhibit distinct molecular consequences
PLOS Genetics: published September 11, 2020 | https://doi.org/10.1371/journal.pgen.1009019
Using prior information from humans to prioritize genes and gene-associated variants for complex traits in livestock
PLOS Genetics: published September 14, 2020 | https://doi.org/10.1371/journal.pgen.1008780
ZNF423 patient variants, truncations, and in-frame deletions in mice define an allele-dependent range of midline brain abnormalities
PLOS Genetics: published September 14, 2020 | https://doi.org/10.1371/journal.pgen.1009017
The causal effect of obesity on prediabetes and insulin resistance reveals the important role of adipose tissue in insulin resistance
PLOS Genetics: published September 14, 2020 | https://doi.org/10.1371/journal.pgen.1009018
The Arabidopsis PHD-finger protein EDM2 has multiple roles in balancing NLR immune receptor gene expression
PLOS Genetics: published September 14, 2020 | https://doi.org/10.1371/journal.pgen.1008993
A Novel Recessive Mutation in SPEG Causes Early Onset Dilated Cardiomyopathy
PLOS Genetics: published September 14, 2020 | https://doi.org/10.1371/journal.pgen.1009000
TENET 2.0: Identification of key transcriptional regulators and enhancers in lung adenocarcinoma
PLOS Genetics: published September 14, 2020 | https://doi.org/10.1371/journal.pgen.1009023
Meiotic cohesins mediate initial loading of HORMAD1 to the chromosomes and coordinate SC formation during meiotic prophase
PLOS Genetics: published September 15, 2020 | https://doi.org/10.1371/journal.pgen.1009048
Polygenic risk for autism spectrum disorder associates with anger recognition in a neurodevelopment-focused phenome-wide scan of unaffected youths from a population-based cohort
PLOS Genetics: published September 17, 2020 | https://doi.org/10.1371/journal.pgen.1009036
PIG-1 MELK-dependent phosphorylation of nonmuscle myosin II promotes apoptosis through CES-1 Snail partitioning
PLOS Genetics: published September 18, 2020 | https://doi.org/10.1371/journal.pgen.1008912
Leveraging correlations between variants in polygenic risk scores to detect heterogeneity in GWAS cohorts
PLOS Genetics: published September 21, 2020 | https://doi.org/10.1371/journal.pgen.1009015
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Candidate variants in TUB are associated with familial tremor
PLOS Genetics: published September 21, 2020 | https://doi.org/10.1371/journal.pgen.1009010
Coordinate genomic association of transcription factors controlled by an imported quorum sensing peptide in Cryptococcus neoformans
PLOS Genetics: published September 21, 2020 | https://doi.org/10.1371/journal.pgen.1008744
AMP1 and CYP78A5/7 act through a common pathway to govern cell fate maintenance in Arabidopsis thaliana
PLOS Genetics: published September 22, 2020 | https://doi.org/10.1371/journal.pgen.1009043
Prioritizing sequence variants in conserved non-coding elements in the chicken genome using chCADD
PLOS Genetics: published September 23, 2020 | https://doi.org/10.1371/journal.pgen.1009027
Aldh inhibitor restores auditory function in a mouse model of human deafness
PLOS Genetics: published September 24, 2020 | https://doi.org/10.1371/journal.pgen.1009040
Deficiency of the Tbc1d21 gene causes male infertility with morphological abnormalities of the sperm mitochondria and flagellum in mice
PLOS Genetics: published September 25, 2020 | https://doi.org/10.1371/journal.pgen.1009020
Restriction on self-renewing asymmetric division is coupled to terminal asymmetric division in the Drosophila CNS
PLOS Genetics: published September 28, 2020 | https://doi.org/10.1371/journal.pgen.1009011
Imaginal disc growth factor maintains cuticle structure and controls melanization in the spot pattern formation of Bombyx mori
PLOS Genetics: published September 28, 2020 | https://doi.org/10.1371/journal.pgen.1008980
Biological insights from multi-omic analysis of 31 genomic risk loci for adult hearing difficulty
PLOS Genetics: published September 28, 2020 | https://doi.org/10.1371/journal.pgen.1009025
Alleviating chronic ER stress by p38-Ire1-Xbp1 pathway and insulin-associated autophagy in C. elegans neurons
PLOS Genetics: published September 28, 2020 | https://doi.org/10.1371/journal.pgen.1008704
Cocoonase is indispensable for Lepidoptera insects breaking the sealed cocoon
PLOS Genetics: published September 28, 2020 | https://doi.org/10.1371/journal.pgen.1009004
A nonsense variant in Rap Guanine Nucleotide Exchange Factor 5 (RAPGEF5) is associated with equine familial isolated hypoparathyroidism in Thoroughbred foals
PLOS Genetics: published September 28, 2020 | https://doi.org/10.1371/journal.pgen.1009028
Snf1 AMPK positively regulates ER-phagy via expression control of Atg39 autophagy receptor in yeast ER stress response
PLOS Genetics: published September 28, 2020 | https://doi.org/10.1371/journal.pgen.1009053
NFIA differentially controls adipogenic and myogenic gene program through distinct pathways to ensure brown and beige adipocyte differentiation
PLOS Genetics: published September 29, 2020 | https://doi.org/10.1371/journal.pgen.1009044
Cis-regulatory differences in isoform expression associate with life history strategy variation in Atlantic salmon
PLOS Genetics: published September 30, 2020 | https://doi.org/10.1371/journal.pgen.1009055
Mutually exclusive dendritic arbors in C. elegans neurons share a common architecture and convergent molecular cues
PLOS Genetics: published September 30, 2020 | https://doi.org/10.1371/journal.pgen.1009029
The STRIPAK signaling complex regulates dephosphorylation of GUL1, an RNA-binding protein that shuttles on endosomes
PLOS Genetics: published September 30, 2020 | https://doi.org/10.1371/journal.pgen.1008819
Correction
Correction: Systems genomics approaches provide new insights into Arabidopsis thaliana root growth regulation under combinatorial mineral nutrient limitation
PLOS Genetics: published September 23, 2020 | https://doi.org/10.1371/journal.pgen.1009107