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. 2020 Dec 4;29(22):3662–3678. doi: 10.1093/hmg/ddaa258

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Wbp11^−/− and Wbp11^+/− embryos and mice do not survive in expected numbers. (A) Number of Wbp11^+/− and Wbp11^+/+ mice observed compared with the expected Mendelian ratio (dotted line) at E17.5 (backcross 2, ^*P = 0.0263), P12 (backcross 2, ^*P = 0.0412) and post-weaning (backcrosses 1 and 2, ^*^*^*^*P = 0.0001). Deviation from expected Mendelian ratios for the Wbp11^+/− × Wbp11^+/+ breeding scheme was calculated by chi-squared test. Dead mice that could not be genotyped were omitted. (B) Percentage of male and female Wbp11^+/− mice that died postnatally. On the left, FVB/N background backcrosses 1–4, ^*^*P = 0.0063. On the right, C57BL/6J background crosses 1–4, ^*^*P = 0.0049. Fisher’s exact test was used to determine significance. (C) Comparison of Wbp11^+/+ and Wbp11^+/− embryo weights at E17.5 normalized to average wildtype body weight (n = 98, ^*^*^*^*P < 0.0001). (D) Photomicrographs and contrast-enhanced micro-computed tomography (micro-CT) scans of E17.5 embryos illustrating the smaller size of Wbp11^+/− embryos and the presence of edema (arrowhead) compared with wildtype embryos from the same litter. (E) Placenta weight of E17.5 embryos (FVB/N) after dissection, normalized to average wildtype placenta weight (n = 98, ^*P = 0.0239). (F) Wbp11^+/− and Wbp11^+/+ E17.5 embryo weights normalized to the weights of their placentas (^*^*^*^*P < 0.0001). (G) Embryo genotypes observed at E8.5 in litters from Wbp11^+/− × Wbp11^+/− matings (backcrosses 2 and 3). Deviation from expected Mendelian ratios (dotted lines) was calculated by chi-squared test (P = 0.0007). (H) Photomicrographs of Wbp11^+/− and wildtype E8.5 embryos. Error bars represent SD. (C, E and F) Unpaired t-test was used to determine significance, and red dots indicate the embryos in (D). Scale bars: 5 mm (D), 500 μm (H).