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. 2019 Jan 11;12(1):dmm036491. doi: 10.1242/dmm.036491

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© 2019. Published by The Company of Biologists Ltd

This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution and reproduction in any medium provided that the original work is properly attributed.

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Fig. 5. — Reduced Raldh2 gene dosage results in PAA and aortic arch abnormalities in heterozygous Hectd1^opm/+ mutant embryos. (A-H) Intracardiac ink injections of E10.5 (32- to 36-somite; A-D) and E17.5 (E-H) embryos to visualize the PAAs and aortic arch organization. (A-D) Right-sided views of E10.5 wild-type (Hectd1^+/+;Raldh2^+/+; A), Hectd1^+/+;Raldh2^+/− (B), Hectd1^+/opm;Raldh2^+/+ (C) and Hectd1^+/+;Raldh2^+/+ (D) embryos, showing the 3rd, 4th and 6th PAAs, with examples of hypoplastic 4th PAAs (arrows). Few wild-type (n=3/16, 19%), Hectd1^+/+;Raldh2^+/– (n=3/11, 27%) and Hectd1^opm/+;Raldh2^+/+ (5/12, 42%) embryos showed hypoplastic 4th PAAs (Table 3). On the other hand, the 4th PAA was hypoplastic in two-thirds of double heterozygous Hectd1^opm/+;Raldh2^+/– embryos (n=11/16, 69%, P<0.0001, compared with wild type by Chi² test). (E-H) At E17.5, 70% (7/10) of Raldh2 heterozygotes (F) and 71% (5/7) Hectd1 heterozygotes demonstrate similar length of the transverse arch between the brachiocephalic and left common carotid arteries (brackets) compared with wild type (E). This segment is shortened in Hectd1^opm/+;Raldh2^+/– double heterozygotes (H, asterisk), resulting in a shortened transverse arch between the brachiocephalic and left common carotid arteries in over half of embryos examined (4/7, 57%; P<0.05, Chi² test). Scale bars: 50 µm (D) and 250 µm (H). AA, ascending aorta; DA, ductus arteriosus; DAo, descending aorta; LCC, left common carotid; LSA, left subclavian artery; PT, pulmonary trunk; RCC, right common carotid; RSA, right subclavian artery.