Cited by 379Hereditary Neurological DisordersAmyotrophic Lateral Sclerosis ResearchMicrotubule and mitosis dynamics

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Kinesin Mutations Cause Motor Neuron Disease Phenotypes by Disrupting Fast Axonal Transport in Drosophila

Hurd, Daryl D, Saxton, William M·1996·Genetics

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Abstract

Previous work has shown that mutation of the gene that encodes the microtubule motor subunit kinesin heavy chain (Khc) in Drosophila inhibits neuronal sodium channel activity, action potentials and neurotransmitter secretion. These physiological defects cause progressive distal paralysis in larvae. To identify the cellular defects that cause these phenotypes, larval nerves were studied by light and electron microscopy. The axons of Khc mutants develop dramatic focal swellings along their lengths. The swellings are packed with fast axonal transport cargoes including vesicles, synaptic membrane proteins, mitochondria and prelysosomal organelles, but not with slow axonal transport cargoes such as cytoskeletal elements. Khc mutations also impair the development of larval motor axon terminals, causing dystrophic morphology and marked reductions in synaptic bouton numbers. These observations suggest that as the concentration of maternally provided wild-type KHC decreases, axonal organelles transported by kinesin periodically stall. This causes organelle jams that disrupt retrograde as well as anterograde fast axonal transport, leading to defective action potentials, dystrophic terminals, reduced transmitter secretion and progressive distal paralysis. These phenotypes parallel the pathologies of some vertebrate motor neuron diseases, including some forms of amyotrophic lateral sclerosis (ALS), and suggest that impaired fast axonal transport is a key element in those diseases.

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Authors: Hurd, D. D., & Saxton, W. M.
Affiliations: Indiana University Bloomington (US)
Corresponding author: Saxton, William M
Venue: Genetics
Publisher: Oxford University Press (OUP)
Published: 1996-11-01
Volume: 144
Issue: 3
Pages: 1075-1085
ISSN: 0016-6731 (ISSN-L)
1943-2631
DOI: 10.1093/genetics/144.3.1075
OpenAlex: W1877780738
Cited by: 379
References: 41
Citations by year: 2026: 1 · 2025: 8 · 2024: 9 · 2023: 14 · 2022: 8 · 2021: 10 · 2020: 8 · 2019: 7 · 2018: 11 · 2017: 16 · 2016: 11 · 2015: 14 · 2014: 18 · 2013: 18 · 2012: 22
FWCI: 3.52
Type: journal article
Language: en
Open access: closed
Licence: academic.oup.com/journals/pages/open_access/funder_policies/chorus/standard_publication_model (vor)
Locations: Genetics (publishedVersion)
PubMed (publishedVersion)
Funders: American Heart Association
National Institutes of Health (5t32gm007227-21)
National Institute of General Medical Sciences (T32GM-07227, R01GM046295, GM-46295)
Topics: Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Hereditary Neurological Disorders
Health Sciences › Medicine › Clinical Neurology › Amyotrophic Lateral Sclerosis Research
Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Microtubule and mitosis dynamics
Keywords: kinesin heavy chain, motor neuron disease, fast axonal transport, retrograde transport, anterograde transport, axonal swellings, organelle transport, synaptic boutons, neurotransmitter release, amyotrophic lateral sclerosis, Drosophila
MeSH terms: Animals, Axons, Biological Transport, Calcium-Binding Proteins, Drosophila, Female, Male, Membrane Glycoproteins, Membrane Proteins, Nerve Tissue Proteins, Paralysis, Phenotype, Rabbits, Organelles, Cell Adhesion Molecules, Neuronal, Mutagenesis, Motor Neuron Disease, Kinesins, Presynaptic Terminals, Animals, Genetically Modified, Qa-SNARE Proteins, Synaptotagmins, HSP40 Heat-Shock Proteins, Mice

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Kinesin Mutations Cause Motor Neuron Disease Phenotypes by Disrupting Fast Axonal Transport in Drosophila

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