BACKGROUND Neurons are polarized cells with extreme geometries. Multiple dendrites
and one axon generally emerge from a single cell body and establish synaptic contacts with …

Axonal transport is essential for neuronal function, and many neurodevelopmental and
neurodegenerative diseases result from mutations in the axonal transport machinery …

Cytoplasmic dynein is an approximately 1.4 MDa multi‐protein complex that transports many
cellular cargoes towards the minus ends of microtubules. Several in vitro studies of …

Charcot-Marie-Tooth (CMT) neuropathies comprise a group of monogenic disorders
affecting the peripheral nervous system. CMT is characterized by a clinically and genetically …

Until recently, dynein was the least understood of the cytoskeletal motors. However, a wealth
of new structural, mechanistic, and cell biological data is shedding light on how this …

Homozygous SMN1 loss causes spinal muscular atrophy (SMA), the most common lethal
genetic childhood motor neuron disease. SMN1 encodes SMN, a ubiquitous housekeeping …

Motor neuron diseases (MNDs) are an etiologically heterogeneous group of disorders of
neurodegenerative origin, which result in degeneration of lower (LMNs) and/or upper motor …

Hereditary spastic paraplegias are heterogeneous neurodegenerative disorders
characterized by progressive spasticity of the lower limbs due to degeneration of the …

Inherited peripheral neuropathies are a genetically and phenotypically diverse group of
disorders that lead to degeneration of peripheral neurons with resulting sensory and motor …

Mutations in the human DYNC1H1 gene are associated with neurological diseases.
DYNC1H1 encodes the heavy chain of cytoplasmic dynein-1, a 1.4-MDa motor complex that …