Skip to main content
. Author manuscript; available in PMC: 2020 Jul 22.
Published in final edited form as: Dev Cell. 2019 Jun 6;50(2):212–228.e6. doi: 10.1016/j.devcel.2019.05.015

Figure 2. Activation of p53 in the neural crest leads to diverse developmental defects that depend on the degree and timing of p53 activation.

Figure 2

(A) (i-ii) Wnt1-Cre;Trp53LSL−25,26,53,54/+ mice display small belly spots (arrow, 84%, n=13) and have slightly shortened snouts. (iii-v) Alizarin red (bone) staining on P21 skulls and quantification (mean±SD) of bone length (n=7 per genotype). Dotted lines outline the mesodermally-derived parietal bones (p) and the NCC-derived frontal bones (f), nasal bones (n) and mandibles (m). *p<0.05. (vi-viii) P21 Wnt1-Cre;Mdm2flox/+;Mdm4+/− mice display large white belly spots (arrow, 100%, n=9) and have short snouts.

(B) (i-iv) E12.5–13.5 Wnt1-Cre;Trp53LSL−25,26/+ embryos display cleft face (arrows i-iii, 36%, n=11) and have short snouts (iv, 100%, n=11). (v-viii) A single Wnt1-Cre;Trp53LSL−25,26/+ embryo that was viable at E15.5 (v, n=1) had cleft palate (*, vi), small outer ears (vii) and hypoplastic nasal passages (viii). (ix-x) E13.5 Wnt1-Cre;Trp53LSL−25,26/+ embryos display persistent truncus arteriosus (PTA, ix, 100%, n=6) and ventricular septal defects (VSD, x, 100%, n=3).

(C) Wnt1-Cre;Mdm2flox/flox embryos display an absence of frontonasal tissue (i, 100%, n=17), exencephaly (Ex, 47%, n=17), and persistent truncus arteriosus (PTA, ii, 100%, n=4).

(D) (i) Sox10-Cre;Trp53LSL−25,26,53,54/+ mice do not have discernable belly spots (100%, n=5). (ii) Sox10-Cre;Mdm2flox/+;Mdm4+/− mice display small white belly spots (arrow, 67%, n=3).

(E) Sox10-Cre;Trp53LSL−25,26/+ embryos display short snouts (i, 100%, n=9), small outer ears (dotted outline, ii, 100%, n=9), cleft palate (*, iii, 100%, n=4) and normally septated heart outflow tracts (iv, 100%, n=3). (F) Sox10-Cre;Mdm2flox/flox embryos display a severe loss of frontonasal tissue (i-ii, 63%, n=8) and have normally septated heart outflow tracts (iii, 100%, n=5).

Ao: aorta. PA: pulmonary artery. Scale bar: 5mm (A,D), 2mm (Bi,ii,iv,v,vii), 1mm (Ci;Ei,ii;Fi,ii), 400µm (Biii,vi,viii,x;Cii;Eiii,iv;Fiii). Sectioning plane: coronal (Biii,vi,viii;Eiii), transverse (Bx;Cii;Eiv;Fiii). See also Figure S1, Figure S2, Table S1.