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Fig. 2. Pds5B deficiency results in growth retardation, abnormal
skeletal patterning and cleft palate. (A) The weight of
Pds5B-/- P0 mice is lower than that of wild-type
littermates (WT, n=43 and Pds5B-/- mice,
n=35). Error bars represent s.e.m. *P<0.001,
Student's unpaired t-test. (B,C) Morphology of wild-type and
mutant mice. (B) Note short stature, facial dysmorphism, short limbs (arrow),
short snout (arrowhead), and small head in Pds5B-/-
compared to wild-type P0 mice. (C) Short mandible (arrow) in
Pds5B-/- neonate (right) compared to wild-type (left).
(D-I) Alizarin Red S and Alcian Blue staining of newborn mouse
skeleton. Red indicates bone and blue indicates cartilage. (D) Mandibles; (E)
ulna, radius and humerus of forelimbs; (F) scapula of forelimbs. Bone lengths
(square bracket) are indicated by numbers (lengths in mm ± s.d.). Note
the length of mandible, ulna, radius and humerus is significantly shorter in
Pds5B-/- compared to wild-type. Sample sizes for each
genotype range from 18 to 34. P<0.001, Student's unpaired
t-test. (G) Sternum. Arrows, unfused ossification centers;
arrowheads, delayed or missing ossification centers. (H) Ribs. The arrow
points to the C7-T1 fusion. (I) Thoracic (T) and lumbar (L) vertebrae. The
arrow points to the unfused L3 vertebra and arrowheads point to the
hypoplastic 13th ribs in Pds5B-/- neonate. (J,K)
Complete cleft palate (arrow) and thin upper-lip (arrowhead) of a
Pds5B-/- neonate (K) compared with wild-type control (J).
(L,M) Alizarin Red S and Alcian Blue staining of neonate skulls. The
edges of palatal bones are marked by dashed lines. Posterior parts of the
palatal bones in Pds5B-/- mutant failed to meet at the
midline (arrows, M), compared with wild-type littermate (L). (N-S)
Palatogenesis defects illustrated using H&E stained coronal sections of
wild-type and Pds5B-/- E12.5, E14.5, and E18.5 embryos.
Wild-type (N-P); Pds5B-/- (Q-S). ps, palatal shelf; n,
nasal bone; to, tongue. Arrow in S points to the unfused palatal shelves.
Scale bars:1 mm.
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