The N-terminal calponin homology domain of IFT81 interacts with the N-terminal domain of IFT72 to form a tubulin binding module required for transportation of tubulin during ciliogenesis.8,19,20 Like other IFT-B core members, mutations in IFT81 have been observed to cause syndromic ciliopathies featuring renal medullary cysts, paraxial polydactyly, early onset rod-cone dystrophy, cerebellar atrophy, and intellectual disability.21 However, thus far, the association between mutations in IFT81 and nonsyndromic human disease has not been reported. This evidence concerns the gene IFT81 and ciliopathy.