Very recently, new craniofacial phenotypes, i.e., metopic craniosynostosis (CS) and cleft lip and palate, were reported in two individuals carrying heterozygous variants–c.2466+1G>A in intron 24 and c.2333C>A in exon 23 of EFTUD2. These cases were initially suspected of having an alternative craniofacial syndrome and were eventually diagnosed with MFDGA (Lacour et al., 2019). Here, EFTUD2 is linked to metopic craniosynostosis.