DYNC1H1 is known to bind both bicd cargo adaptor (BICD2), a microtubule motor adaptor associated with SMA lower extremity–predominant 2, and lissencephaly 1 (LIS1), a dynein regulator associated with MCD/lissencephaly (18), which might explain why the variant of DYNC1H1 could lead to pachygyria or MCD. Here, DYNC1H1 is linked to lissencephaly spectrum disorders.