Notably, human Drp1 mutations also lead to neurodevelopmental defects with post-neonatal lethality, developmental delay, late-onset neurological decline, or optic atrophy (Fahrner et al., 2016; Gerber et al., 2017; Vanstone et al., 2016; Waterham et al., 2007); however, our current understanding of Drp1’s function outside of mitochondrial division is limited. Here, DNM1L is linked to Global developmental delay.