Deletion mutations that affect the ufm‐1 cascade result in reduced fecundity and life span in worms (Hertel et al., 2013)RNAi knockdown against Ufm1 decreases life span and causes locomotive defects in fruit flies (Duan et al., 2016)A homozygous mutation in UFM1 causes early‐onset encephalopathy with progressive microcephaly in humans (Nahorski et al., 2018). Here, UFM1 is linked to microcephaly.