Figures
Sensitivity of mouse brain development to Zfp423 mutations.
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
Citation: (2020) PLoS Genetics Issue Image | Vol. 16(9) October 2020. PLoS Genet 16(9): ev16.i09. https://doi.org/10.1371/image.pgen.v16.i09
Published: October 1, 2020
Copyright: © 2020 . This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
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.
Download September's cover page.
Image Credit: Ojas Deshpande