3‐M syndrome has been causally related to CUL7, OBSL1, and CCDC8 variants, but the underlying mechanisms remain largely unknown.1, 6, 17 Huber et al reported for the first time that CUL7 variants are convincingly associated with 3‐M syndrome in 2005. This evidence concerns the gene OBSL1 and multiple congenital anomalies-hypotonia-seizures syndrome 3.