Total body cep164 deficiency is embryonic lethal in mice, associated with holoprosencephaly, abnormal heart development, and gross patterning defects, and conditional cep164 deficiency in multi‐ciliated cells causes hydrocephalus, lung disease, and infertility, supporting a role for CEP164 in ciliary function (Siller et al., 2017). This evidence concerns the gene CEP164 and holoprosencephaly.