SPDL1 (spindle apparatus coiled-coil protein 1) encodes the hSpindly protein, which serves as a critical regulator of cell cycle progression and mitotic checkpoint control. The protein functions as an adapter linking dynein motor complexes to various cellular cargos and converts dynein from non-processive to highly processive in the presence of dynactin 1. SPDL1 plays essential roles in maintaining spindle checkpoint silencing during mitosis by coordinating microtubule attachment through kinesin recruitment and mitotic checkpoint signaling 1. Beyond its mitotic functions, SPDL1 is involved in cell migration processes. Disease relevance includes associations with abnormal neurological development, as mutations in SPDL1 have been identified in consanguineous families with neurogenetic disorders through whole-exome sequencing studies 2. The protein's aberrant expression is also linked to pulmonary interstitial fibrosis and malignant tumor development 1. Clinically, SPDL1 dysfunction contributes to various pathological conditions, particularly neurological disorders where it represents one of 33 newly identified recessive disease genes affecting brain function 2. The identification of SPDL1 mutations expands our understanding of neurogenetic disease mechanisms and provides potential targets for therapeutic intervention.