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. 2011 Feb 11;88(2):150–161. doi: 10.1016/j.ajhg.2011.01.003

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© 2011 The American Society of Human Genetics. Published by Elsevier Ltd. All right reserved.

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faf1 Expression Studies and Pharyngeal Arch Development in Zebrafish Larvae

(A–D) Expression pattern of faf1 in control zebrafish of different stages is shown by whole-mount in situ hybridization. At 24 hpf and 30 hpf, strong faf1 expression in pharyngeal arch primordia (PP) (A and B). Dorsal views of the pharyngeal region are shown; the rostral side is down. At 56 hpf, there is strong faf1 expression in all arches, and the most prominent expression is in the first (mandibular, M) and second (hyoid, H) arches (C and D). Scale bars represent 100 μm.

(E–L) Ventral and side views of Alcian-blue-stained 5 dpf wild-type (E, G, I, and K) and faf1^KD (F, H, J, and L) larvae; the focus is on pharyngeal cartilage. At 6 ng/embryo, all cartilages are generally smaller, ceratohyal cartilage is oriented more medially (black arrows), branchial cartilages are partially missing (black asterisk), and Meckel's cartilage is oriented ventrally (double black arrow), resulting in an open-mouth-like appearance. Abbreviations for cartilege are as follows: bb, basibranchial; bh, basihyal; cb, ceratobranchial; ch, ceratohyal; ep, ethmoid plate; hb, hypobranchial; hs, hyosymplectic; m, Meckel's; pch, parachordal; pq, palatoquadrate; and tr, trabecula. Scale bars represent 200 μm.

(M and N) Macroscopic side views of the head of 5 dpf control (M) and faf1^KD (N) zebrafish embryos. Faf1^KD embryos have an open-mouth-like appearance (white arrow). Scale bars represent 200 μm.

(O and P) Green fluorescent protein under fli1 promoter in 2 dpf Tg(fli1:eGFP)^y1 wild-type (O) and faf1^KDp53^KD (P) zebrafish larvae. The CNC of pharyngeal arches defective in faf1^KDp53^KD zebrafish larvae is shown. Scale bars represent 50 μm.

(Q–T) sox9a (Q and R) and col2a1 (S and T) expression in wild-type (Q and S) and faf1^KDp53^KD (R and T) larvae. Sox9a expression is reduced in pharyngeal arches of 30 hpf faf1^KDp53^KD larvae (black arrowheads); a dorsal view is shown. Col2a1 expression is absent in the mandible of 2 dpf faf1^KDp53^KD larvae (black arrowhead); a dorsal view is shown. Scale bars represent 100 μm.