Figure 6.

Frequency of destabilized individual axon growth varies in register with changes in RA-mediated patterning in a series of 22q11.2 gene-related mutant axons. (A) 2D renderings of individual CN V sensory neurons and their distal and proximal axons (top), and CN V motor neurons and their distal axons (bottom) from Tbx1^+/− mutant. (B) Frequency of branching, misrouting and sprouting CN V axon phenotypes in Tbx1^+/− compared with WT. (C) CN V axon tortuosity in Tbx1^+/−. (D) Comparison of number of phenotypes/axon in Tbx1^+/− versus WT embryos. Numbers of axons analyzed in each category are given to the right of the graphic record. (E) Individual CN axon growth in Tbx1^+/− embryos resembles that in WT, as does hindbrain A–P patterning. (F) 2D renderings of Ranbp1^−/− CN V sensory neurons and their proximal and distal axons (top) and confocal image of Ranbp1^−/− CN V motor neurons and their distal axons (bottom). (G) Frequency of branching, misrouting and sprouting CN V axon phenotypes in Ranbp1^−/− and Ranbp1^+/− compared with WT controls. (H) CN V axon tortuosity in Ranbp1^−/− compared with WT. (I) Comparison of number of phenotypes/axon in Ranbp1^−/−, Ranbp1^+/− and WT. (J) Growth and trajectories of individual Ranbp1^−/− axons are similar, but apparently more frequent and severe than those in LgDel^+/−, in register with similar but more substantial changes in A–P hindbrain patterning. (K) Growth and trajectory of individual Ranbp1^+/− axons is similar to WT, as is hindbrain A–P patterning. (L) 2D renderings of CN V motor neurons (circles) and sensory neurons (arrows) in LgDel^+/−:Raldh2^+/−. (M) Frequency of branching, misrouting and sprouting in LgDel^+/−:Raldh2^+/− versus WT CN V axons. (N) CN V axon tortuosity in LgDel^+/−:Raldh2^+/−. (O) Comparison of number of phenotypes/axon in LgDel^+/−:Raldh2^+/− versus WT. (P) Growth and trajectory of individual LgDel^+/−: Raldh2^+/− axons is similar to WT, in parallel with the rescue of hindbrain A–P patterning toward WT.