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Fig. 2. Skeletal abnormalities in Prx1-Hif1
mutants.
(A-C) Alizarin Red and Alcian Blue staining of wild-type (top) and
Prx1-Hif1
(bottom) mouse forelimbs. E18.5 littermates
demonstrate severe retardation in limb skeleton formation (A) and joint fusion
(B). Autopod skeletons (C) show cartilage formation at the periphery of the
forming digits and joint loss in the Prx1-Cre-Hif1
limb. (D) Histological sections of E13.5 control (top) and
Prx1-Cre-Hif1
(bottom) elbows. In the
Prx1-Cre-Hif1
joint, the presence of apoptotic cells
with shrunken cytoplasm and condensed nuclei is observed. (E,F)
Histological sections through the digits revealed partial joint cavitation (E)
and loss of phalanx (F). (G-I) MicroCT analysis of 21-day-old
Prx1-Cre-Hif1
and WT autopod: dorsal view
demonstrates severe retardation in the Prx1-Cre-Hif1
skeleton including bone deformation, lack of phalanges (bracket), and joint
fusion. H and I show dorsal and lateral views, respectively, of digit 3.
Yellow arrows indicate fused joints and fused sesamoid bones.