Dyneins are a class of microtubule (MT)-based motors that generate force and motility toward the minus-end of MTs by converting chemical energy in the form of adenosine triphosphate (ATP) to mechanical work. Dyneins are involved in a variety of cellular processes, including intracellular transport, mitosis, and ciliary motility. Dyneins are repurposed for accomplishing these tasks through the recruitment of a variety of accessory proteins, that regulate the assembly, activation, and recruitment of the dynein complex to its specific cellular targets. Consistent with its fundamental roles in neurobiology and cell development, mutations in dynein and dyneinassociated factors are linked to ciliopathies and neurodegenerative diseases.