Reduced and delayed craniofacial skeletogenesis in Tmem107–/– embryos. Micro–computed
tomography (CT) analysis of Tmem107–/– animals at E15.5 (A–F).
Lateral view shows altered morphology and level of mineralization in
maxillary, mandibular, and parietal bones in Tmem107–/– mutants and absence of the
premaxillary bone (A, B). Ventral view reveals deformed mandibular bone
and shorter frontal and parietal bones in Tmem107–/– embryos (D) compared to
wild-type (wt) (C). On frontal view, mandibular bones have a different
shape and premaxillary bone is not well mineralized in Tmem107–/– mutants (F) in contrast
to wt animals (E). Alcian blue/Alizarin red staining at E16.5
demonstrates delayed mineralization and altered morphology of frontal,
parietal, and premaxillary bones in Tmem107–/– embryos (H,
J) in comparison to wt (G,
I). Palatine bones and palatal process of the maxillary
bone are reduced in Tmem107–/–
mutants (L) compared to wt (K). Mandibular
bones are deformed in Tmem107–/– embryos (N) in contrast
to wt animals (M). SOX9 expression in developing cartilages
of interorbital septum and surrounding vomeronasal organ in wt animals
through the palate development (O–R). Pattern of SOX9
expression is altered in Tmem107–/– mutants in correspondence to
cartilage phenotype (S–V). Cartilages surrounding the
vomeronasal organ exhibit altered shape; they were fused in midline
(S′, T′, U′, V′)
or missing (yellow arrow in T, T′). Nuclei are
counterstained by DAPI (blue). fr, frontal bone; md, mandibular bone;
mx, maxillary bone; ns, nasal septum; pl, palatal bone; pmx,
premaxillary bone; pr, parietal bone; t, temporal bone; vnc, vomeronasal
cartilage; vno, vomeronasal organ. Scale bar = 100 µm.