We generated Fam83h-deficient mice (Fam83hem2(IMPC)Ccpcz, Fam83h-/-) and mice lacking a part of N-terminal CK1-binding domain (Fam83h∆87/∆87). Co-IP with anti-FAM83H antibody has confirmed the FAM83H–CK1 interaction in Fam83hwt/wt samples, which was absent in Fam83h-/- lysates. Additionally, samples from Fam83hΔ87/Δ87 mice were less likely to show FAM83H–CK1 interaction. Similarly, when CK1a antibody was used for pull downs, FAM83H–CK1 interaction was observed in Fam83hwt/wt lysates , but not in Fam83h-/- and Fam83hΔ87/Δ87 samples.