Recently, we have identified mutations in PRDM15 that can cause typical GAMOS renal malformations in embryos of the South African clawed frog Xenopus laevis (X. laevis) (Mann et al., 2021) and demonstrated that genetic deletion of Prdm15 in mice recapitulates brain malformations observed in syndromic HPE (GAMOS) patients (Mzoughi et al., 2020). Here, PRDM15 is linked to holoprosencephaly.