Figure 2.
Loss of Akirin2 in the limb epithelium leads to soft-tissue syndactyly. (a) Interdigital tissue regression is impaired in Akirin2 Emx-KO forelimb (digits 2/3) and hindlimb (digits 2/3/4). (b) Alcian Blue/Alizarin Red staining at P0 and in one of the few surviving mutants at P10 shows that the Akirin2 Emx-KO limb does not have fused bone or cartilage elements, though broader Alizarin Red (bone) staining is observed in the phalanges (red arrows). (c) A very rare Akirin2 Emx-KO that survived until P30 contains clear syndactyly of the mature forelimb and hindlimb. (d–g) Quantification of interdigital tissue regression shows impaired tissue regression between digits 2–3 of the forelimb (d) and 2–4 of the hindlimb (f,g) in Akirin2 Emx-KO mice. The difference in forelimb interdigital regression between digits 3–4 approaches statistical significance (e; p = 0.054). n = 3 animals/genotype for E12 & E13; 4 animals/genotype for E14, *p < 0.05, **p < 0.01, ***p < 0.001, ****p < 0.0001, t-test. (h) Immunostaining of Akirin2 Emx-KO distal limb buds at E12.5 shows that Akirin2 expression is extensively lost in the interdigital space between digits 2–3 in both fore- and hindlimb. In the forelimb, most cells between digits 3–4 remain Akirin2-positive, while in the hindlimb more Akirin2-negative cells are observed (interdigital regions indicated between arrowheads). Scale bar: 1 mm in a-c; 50 μm in h.