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. 2020 Jan 10;11:195. doi: 10.1038/s41467-019-14077-2

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© The Author(s) 2020

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/.

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Fig. 3 — Analysis of Cdkn1c-MADM-7 (MADM-7^GT/TG,Cdkn1c;Emx1^Cre/+) with maternal deletion (a–f), Cdkn1c-MADM-7 (MADM-7^GT/TG,Cdkn1c;Emx1^Cre/+) with paternal deletion (g–l), and cKO-Cdkn1c-MADM-7 (MADM-7^{GT,Cdkn1c/TG,Cdkn1c};Emx1^Cre/+) (m–r). The parent from which the MADM cassettes with or without recombined Cdkn1c-flox allele (Cdkn1c) were inherited is indicated in the respective genotypes in pink (mother) and blue (father). Predicted Cdkn1c expression in GFP⁺ (green), tdT⁺ (red), GFP⁺/tdT⁺ (yellow) and unlabeled (gray) MADM-labeled cells (a, g, m); and true relative levels of Cdkn1c expression in GFP⁺ (green bar), tdT⁺, (red bar), GFP⁺/tdT⁺ (yellow bar) MADM-labeled cells (b, h, n) in Cdkn1c-MADM-7 with maternal deletion (a, b), Cdkn1c-MADM-7 with paternal deletion (g, h), and cKO-Cdkn1c-MADM-7 (m, n) at E16 are indicated (MM, matUPD; PP, patUPD; MP, control). Data points indicate individual animals (n = 5–7).Outlier values are not shown (see Source Data File). Bars indicate median. Imprinting of Cdkn1c (arrow, expression; ball on stick, repression) and predicted expression (0×, 1×, 2×) is indicated. Parental origin of chromosome is indicated (M, maternal; P, paternal) and conditional deletion of Cdkn1c is marked with red cross. MADM-labeling (GFP⁺, green; tdT⁺, red; GFP⁺/tdT⁺, yellow) (c, d, i, j, o, p), cortical width (μm) is shown with an error bar representing SEM (e, k, q), and G/R ratio (geometric mean) of single MADM-labeled cortical projection neurons (f, l, r) in Cdkn1c-MADM-7 with maternal deletion (c–f), Cdkn1c-MADM-7 with paternal deletion (i–l), and cKO-Cdkn1c-MADM-7 (o–r) at P21 are indicated. Boxed areas in overview images (c, i, o) show representative images of the extent of microcephaly at higher resolution (d, j, p). Nuclei were stained using DAPI (blue). Scale bar, 500 μm (overview (c, i, o)) and 90 μm (inset (d, j, p)). Note the decreased numbers of Cdkn1c^−/− cells when compared to Cdkn1c^+/+ cells in (f) and (l) but equipotency/equal numbers of Cdkn1c^−/− cells in (r) regardless of the UPD status. Data points (e, f, k, l, q, r) indicating individual animals (n = 3). NCX neocortex, HC hippocampus. Source data are provided as a Source Data file.