Figure 1. Tongue phenotypes in Ofd1 mutant mice.

(A, B, E–H) Frontal sections showing histological images in wild-type (A, E, G), Ofd1^fl;Wnt1Cre(HM) (B) and Ofd1^fl/WT;Wnt1Cre(HET) (F–F’’, H) at embryonic day (E) 18.5. Arrow indicating sparse tissue (B). *: tongue (A). Green, blue, and yellow arrows indicating normal muscle, ectopic sparse tissue, and ectopic bone, respectively (F). F’ and F’’ are high magnification of F indicated by blue and yellow arrow, respectively. Arrows indicating tongue frenum region (G, H). The presence of clefts and multiple protrusions; n=58/58. (C, D) Image showing oral view of tongue in wild-type (C) and Ofd1^fl/WT;Wnt1Cre(HET) (D). Arrowhead and arrow indicating ectopic protrusion and cleft, respectively (D). Lack of tongue frenum in Ofd1^fl/WT;Wnt1Cre(HET); n=30/58. Scale bars: 200 µm (A, B, E, F, G, H), 100 µm (F', F'').

Figure 1—figure supplement 1. Abnormal shaped tongue in Ofd1^fl/WT;Wnt1Cre(HET) mice.

Oral and frontal view of normal tongue in wild-type mice and abnormal shaped tongue in Ofd1^fl/WT;Wnt1Cre(HET) mice.

Figure 1—figure supplement 2. Bone in tongue of Ofd1^fl/WT;Wnt1Cre(HET) mice.

Frontal sections showing in situ hybridization of Runx2 (A, B) and histology (C, D) in wild-type (A, C) and Ofd1^fl/WT;Wnt1Cre(HET) (B, D). Arrow and arrowhead indicating bone region and connection between bone in the tongue and mandibular bone, respectively (B). Arrowheads and arrow indicating Meckel’s cartilage and duplicated mandibular bone, respectively (D). Scale bars: 200 µm (A, B), 100 µm (C, D).

Figure 1—figure supplement 3. Tongue phenotypes in Ofd1^fl/WT;Wnt1Cre(HET) mice.

Frontal section showing histological images of tongue in wild-type (A) and Ofd1^fl/WT;Wnt1Cre(HET) mice (B) at embryonic day (E) 18.5. Scale bars: 200 µm (A, B).