SPAG6 directly interacts with CCDC103. (A) Schematic of human SPAG6 and DYNC1H1 proteins. Specific domains identified by Y2H and portion of DYNC1H1 cloned into the LuTHy prey vector are indicated with brackets. The entirety of the SPAG6 CDS was included in the respective LuTHy prey vector. (B) BRET ratios for the interaction between CCDC103 and DYNC1H1 in the presence of DMSO, nocodazole and paclitaxel. (C) BRET ratios for the interaction between CCDC103 and SPAG6 in the presence of DMSO, nocodazole and paclitaxel. (D) Schematic of WT CCDC103 protein and three mutations found in PCD patient mutations. Relative severity of the patient PCD phenotype associated with the mutation indicated in parentheses. (E) BRET ratios for the interaction between DYNC1H1 and WT and mutant CCDC103 proteins. (F) BRET ratios for the interaction between SPAG6 and WT and mutant CCDC103 proteins. **P<0.005, *P<0.05; ns, not significant (two-tailed unpaired Student's t-test).
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