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. Author manuscript; available in PMC: 2017 Aug 23.
Published in final edited form as: Science. 2017 Jun 2;356(6341):eaal3345. doi: 10.1126/science.aal3345

Fig. 5. CR3HD CUX1 promotes chondrogenesis.

Fig. 5

(A) ATDC5 control cells and cells carrying CR3HD CUX1-Ancestral or CUX1-Δ4aa variants were differentiated into chondrocytes. Gene expression levels were measured by RT-qPCR (n=4 biological replicates, each compromising 3 technical replicates) after 7 and 12 days. Error bars indicate standard errors. We used ANOVA and Tukey’s HSD to test for statistical significance. Black stars indicate a significant difference between a CUX1 variant and the control. Red starts indicate a significant difference between CUX1-Ancestral and CUX1-Δ4aa variants. Absence of stars indicates no significant difference. (*) P <0.05, (**) P<0.01, and (***) P<0.001. (B) Proposed mechanism for the reduction of wing size in P. harrisi. The left panel depicts the normal functioning of IHH signaling pathway in vertebrates. CUX1 regulates the expression of cilia related genes such as Ofd1 and promotes chondrogenesis. The right panel depicts the state of the IHH pathway in P. harrisi. Proteins in red have predicted function-altering variants in P. harrisi. We propose that these variants will affect both cilia formation and functioning leading to a reduction in IHH pathway activity. As a result, the pool of proliferating chondrocytes would decrease in wing bones and the number of hypertrophic chondrocytes would increase resulting in impaired bone growth.