Lis1, a protein best known for association with the developmental disease lissencephaly, is an additional dynein regulator that seems to have two functions: Lis1 modulates dynein’s affinity for microtubules in vitro and promotes dynein’s association with dynactin and an activating adaptor (McKenney et al., 2010; Huang et al., 2012; Reiner and Sapir, 2013; Baumbach et al., 2017; DeSantis et al., 2017; Elshenawy et al., 2020; Htet et al., 2020). Here, PAFAH1B1 is linked to lissencephaly spectrum disorders.